Local lipohypertrophy due to recombinant human growth hormone (rhGH) administration is a rare phenomenon. Here, we report a case of an 11-year-old girl who presented with a paraumbilical swelling, approximately one year after the start of rhGH treatment for short stature due to the presumed diagnosis of partial growth hormone insensitivity. Ultrasound imaging revealed an asymmetric distribution of subcutaneous fat tissue at the rhGH administration site, indicating local lipohypertrophy. After sparing her routine injection site and alternating other sites, the swelling disappeared within 6 months. Although the precise cause of local lipohypertrophy resulting from rhGH administration is still unclear, it might be related to the presumed diagnosis of partial growth hormone insensitivity. Human growth hormone (hGH) is usually administered via subcutaneous injections. Besides well-known adverse events such as peripheral edema, benign intracranial hypertension, and slipped capital femoral epiphysis, a less known and rare side effect is local lipohypertrophy, a phenomenon with an incompletely understood pathophysiology. Although innocent and easy to treat, it may give rise to concern in patients and caregivers.An 11-year-old girl was referred to the department of pediatric surgery for a newly developed swelling on the left side of the umbilicus. The swelling varied in size and was painful at times. Yet there was no history of trauma and there were no signs of inflammation or infection. Physical examination revealed a soft, painless swelling on the left side of the umbilicus (2 × 3 cm). There was no sign of a fascia defect and the Valsalva-manoeuvre was negative.The girl had an extensive medical history. Early in life, she suffered from unexplained postnatal hypotonia and respiratory failure, for which extensive investigations were performed. Genetic testing showed a normal female chromosome pattern , and DNA testing for 22q11 microdeletion syndrome, Prader-Willi syndrome, and Steinert syndrome was negative. A cause for these transient postnatal problems could not be identified. In addition, she had a right-sided inguinal hernia at birth and a left-sided inguinal hernia at the age of 10 months; both were surgically corrected. At the age of three years, she underwent surgical correction for an umbilical hernia. Because of short stature due to (presumed) partial growth hormone resistance, she was using rhGH since the age of approximately 10 years. When the girl was 2.5 years old, her growth curve had declined . Her heiWith the aforementioned clinical finding and medical history in mind, the differential diagnosis of the paraumbilical swelling included hernia, local inflammatory/allergic reaction to the medication, lipoma, hemangioma, granuloma, cyst, abscess, and neoplasm. Ultrasound imaging of the swelling was performed, which revealed an asymmetric distribution of subcutaneous fat on the injection site, with a thickness difference of 6 mm (left more than right). There was no sign of hernia. The asymmetric fat distribution at the transjection site led to the suspicion of the rare occurrence of subcutaneous lipohypertrophy. The girl was instructed not to administer rhGH at the site of the swelling anymore and to alternate delivery sites to prevent repeated administration of rhGH at the same site. After 6 months, the swelling had decreased and the pain had disappeared and she had reported no (other) adverse effects.The availability of rhGH has broadened its range of clinical applications . NowadayBecause rhGH is a peptide hormone, it is administered via subcutaneous injections. Studies on the effects of rhGH treatment on lipid metabolism have shown a general lipolytic effect , and locThe pathophysiology of local lipohypertrophy due to rhGH administration is incompletely understood. GH stimulates the production of IGF-1 in adipocyte precursor cells. The effect of IGF-1, however, depends on its local concentration at the level of the adipocyte in the adipose tissue. Low IGF-1 concentrations have a lipolytic effect, whereas high concentrations, when sufficient to activate insulin receptors, can induce intracellular glucose transport and lipogenesis . Similar2 per day  and increased lipogenesis due to higher local IGF-1 concentrations than before initiation of rhGH administration. Unfortunately, proof  of partial GH resistance was not found. In retrospect, the diagnosis of partial GH insensitivity may even be questioned. The rather firm rise of IGF-1 after seven days of treatment with rhGH in a dose of only 0.7 mg per m per day  suggestsIn this 11-year-old girl, rhGH treatment for short stature due to a presumed diagnosis of partial GH resistance caused local, paraumbilical subcutaneous lipohypertrophy. By sparing the swelling and alternating rhGH transjection sites, the lipohypertrophy clearly diminished. Caregivers who treat patients with rhGH should be aware of this rare side effect. Although the precise cause of local lipohypertrophy resulting from rhGH administration is still unclear, it might be related to insensitivity local disequilibrium between rhGH induced lipolysis and IGF-1 concentration dependent lipolysis or lipogenesis.


Spinal cysts are rare findings in veterinary medicine, but they are increasingly recognized due to the availability of advanced imaging techniques. Extradural meningeal cysts in French Bulldogs have not been reported previously and arachnoid cysts (diverticula) have not been reported at the caudal lumbar (L6–L7) region in dogs.Two French Bulldogs, aged 5 and 8 years, were referred for evaluation of lower back pain and bilateral hind limb neurological deficits. Neurologic examination revealed ataxia and postural deficits in both dogs. Magnetic resonance imaging (MRI) showed cauda equina compression due to a cyst-like lesion at the level of L6–L7 in both cases. The dogs underwent dorsal laminectomy and the meningeal cyst was completely removed in one dog and in the other dog the spinal arachnoid diverticula was marsupialized. In Case 1, histopathology of the cysts was performed and MRI was repeated. Both dogs were pain free during follow-up evaluations.Based on radiological, intra-operative and histopathological findings, the first case was diagnosed as a meningocele connected by a pedicle to the caudal tip of the dural sac forming a dural diverticulum categorized as an extradural spinal cyst type Ib, and Case 2 as a type III intradural arachnoid diverticula. It is concluded that spinal cysts should be included in the differential diagnosis of cauda equina syndrome and lower back pain in French Bulldogs. Results of these cases may be useful for diagnostic and treatment management. Cystic-l lining , 4. Intrl lining , 2, 6. MLowrie et al.  introducSpinal cysts and diverticula in humans have been reported to occur primarily in the lumbar spine , 9, wherIn this case report we describe two French Bulldogs, one presenting with a meningeal cyst type Ib and one with a type III  in the caudal lumbar area. The clinical and MRI findings, surgical treatment, histopathology, and follow up are reported.An 8-year-old female French Bulldog weighing 12.4 kg was referred to the Department of Clinical Sciences of Companion Animals at Utrecht University with a history of intermittent faecal incontinence and progressive abnormal gait in the hind limbs since 3 months. Conservative treatment with corticosteroids initially showed improvement but after tapering the dosage, clinical signs recurred. The owners felt that the dog was in pain because they found the dog progressively more depressed/lethargic. On general examination, the dog showed signs related to brachycephalic obstructive airway syndrome. Orthopaedic examination elicited pain on palpation and extension of the L6 to S3 region. Neurological examination identified hind limb ataxia and ambulatory paraparesis and postural deficits in the hind limbs, more severe on the left side. Spinal reflexes were normal in all limbs. The neuroanatomic localization based on the neurological deficits included T3–L3 and based on the lumbosacral pain on palpation and faecal incontinence it was extended until S3. Magnetic resonance imaging  was performed under general anaesthesia of the thoracolumbar, lumbar and lumbosacral spine extending from T6 to Cd3 with sagittal and/or transverse T1-weigted, T2-weighted, T2*-weighted, fluid attenuation inversion recovery (FLAIR) and short tau inversion recovery (STIR) sequences  and butorphanol . Propofol  was used to induce anaesthesia. General anaesthesia was maintained with isoflurane, ketamine  and dexmedetomidine . Medication included cefazolin , buprenorphine , and dexamethasone . A standard dorsal laminectomy from the midpoint of L6 extending over L7 to S1 was performed as described previously , using ain toto, embedded in paraffin, sliced and stained with haematoxylin and eosin (HE) and periodic acid Schiff (PAS). Macroscopically, the cyst was stalked, thin-walled, measured 1.3 × 0.5 cm and filled with clear fluid. Histologically, several optically empty slit-like spaces were lined with cuboidal epithelium and embedded in connective tissue adjacent to a peripheral nerve that showed minor degenerative changes , gabapentin  and prednisolone was tapered down and stopped over a period of a few weeks. The owners were advised to keep the dog quiet and on leash walks for at least 6 weeks. Physical therapy was started at 2 weeks after surgery to support rehabilitation.At 3 months follow-up examination, the dog was without medication and the owners had noted progressive improvement in the clinical signs. The hind limb ataxia was still noticeable, but less severe and the same was true for the faecal incontinence. The dog no longer showed signs of depression/lethargy, ambulatory paraparesis was no longer present, and no pain could be elicited during palpation and/or extension of the lower back. MRI of the thoracic, lumbar and lumbosacral spine was performed, including sagittal and transverse T1W and T2W sequences, transverse T2 * W and sagittal STIR sequences Fig. . The rouA 5-year-old male French Bulldog weighing 10.3 kg was referred to the Department of Clinical Sciences of Companion Animals at Utrecht University with a 3-year history of hind limb ataxia and ambulatory paraparesis without clinical signs of pain. MRI of the cervical and thoracolumbar spine performed 1 year previously in another institution showed intervertebral disc degeneration at multiple levels and mild disc protrusion at T12–T13 but without compression of neural structures. The dog was treated conservatively with corticosteroids without clinical improvement. Ambulatory paraparesis persisted and in addition to the motor deficits, passive urine incontinence developed not related to overflow. Results of initial physical examination were unremarkable; however, a complete neurological examination revealed ataxia with dysmetria and hypermetria of both hind limbs, as well as muscle atrophy and hypotony. The dog had proprioceptive deficits in both hind limbs, but spinal reflexes were normal in all limbs. Based on the neurological deficits, the urinary incontinence, and the results of neurological examination, the neuroanatomic localization was determined as T3–S3.Magnetic resonance imaging was repeated from C1 to S1, including sagittal and transverse T1-weigted and T2-weighted sequences, and a transverse FLAIR sequence of the lumbar spine. Multiple intervertebral discs showed decreased hypointense signal compatible with intervertebral disc degeneration. At the level of C2–C3, T12–T13, T13–L1 and L1–L2 mild disc protrusion was found with visible compression of the ventral subarachnoid space, but without compression or dislocation of the spinal cord. A fusiform cystic lesion dorsal to the cauda equina at the level of L6–L7 was found with an ill-defined cranial and caudal border. The cystic structure was isointense to CSF and compressed the cauda equina Fig. . Based oAnaesthesia and medication for the surgical procedure were identical to Case 1 except that this dog received carprofen  instead of corticosteroids in the postoperative period. The surgical technique was the same as for Case 1. After dorsal laminectomy from halfway L6 over L7 to S1, a dilated dural sac was exposed which was macroscopically identified as SAD. A durotomy was performed over a length of approximately 3 cm, which resulted in free flow of CSF into the spinal canal and collapse of the diverticula. No tissue was available for histological examination. Marsupialization of the SAD was performed by placing interrupted sutures with polydioxanone 5-0 attaching the dural sac to the lateral wall of the laminectomy. Before closure the laminectomy was covered with an autologous fat graft to prevent adhesions.The dog was discharged 1 day after surgery with the following oral medication: amoxicillin/clavulanic acid , tramadol , and carprofen (2 mg/kg q12 h). The owners were advised to keep the dog quiet and on leash walks for 6 weeks.The dog was re-examined at 2 weeks after surgery. The dog showed moderate ataxia and postural reaction deficits in the hind limbs. The dog was both urinary and faecal continent. All spinal reflexes including perineal reflex were normal. Physical therapy and hydrotherapy was started to support rehabilitation. At 6 weeks postoperatively, the motor function of the dog had recovered significantly. Although, the dog was still ataxic, the dog was evidently less paretic in the hind limbs. Postural deficits were improved in both hind limbs and all spinal reflexes were normal. Also, the muscle mass over the hind limbs had progressively increased.In this study, a meningeal cyst and a SAD are reported in the lumbar region of French Bulldogs. Although spinal cysts have been described more frequently in veterinary literature in recent years, prevalence of canine arachnoid diverticula in the caudal lumbar region is very rare , 10. SchCongenital vertebral column deformities like hemivertebrae and degenerative intervertebral disc disease have been reported previously in French Bulldogs and other chondrodystrophic breeds , 18. In Progressive postoperative clinical improvement suggested the causality between the cyst and some of the clinical signs, although this is difficult to confirm due to the absence of a control group. Both cases showed clinical signs consistent with a neuroanatomical localization T3–L3 , and could be further extended until S3 . Persistent neurological deficits, i.e. moderate hind limb ataxia and, although less frequent, persistent intermittent faecal incontinence (Case 1) may be explained by two possible reasons. First, clinical signs of hind limb ataxia may be explained by other detected spinal deformities, i.e. the intramedullary hyperintensity at the level of T12 representing spinal cord oedema, and abnormalities common to the brachycephalic breed, i.e. hemivertebrae, kyphosis, and degenerative disc disease . In a reIn accordance to radiological, intra-operative and histopathological findings our first case was categorized as an extradural meningeal type Ib cyst , 4. TypeIn conclusion, this is the first report of caudal lumbar spinal cysts/diverticula in French Bulldogs and gives us more knowledge and insights into  spinal deformities and abnormalities related to the French Bulldogs breed. Spinal cysts/diverticula should be included in the differential diagnosis of cauda equina syndrome and lower back pain in French Bulldogs. However, further studies are indicated to evaluate the prevalence and possible genetic relation of spinal cysts/diverticula to the French Bulldog breed.


Soft tissue mass following total hip arthroplasty raises several differential diagnoses not limited to infection, hematoma, wear debris, malignancy, and bursitis. Rice body formation in the hip region is an uncommon process denoting a chronic inflammation. We report here the second case of its kind in the medical literature of a wide symptomatic rice-like body cyst complicating a total hip arthroplasty. This is the case of an 82-year-old white female, presenting with a warm, red, and inflated groin five years after revision of right total hip arthroplasty. Surgical intervention reveals a large well circumscribed cyst containing well-organized rice-like bodies. This eventuality was never reported in differential diagnosis of hip periprosthetic soft tissue masses before. This case report helps widening the array of the differential diagnosis in patients presenting with a slow growing soft tissue mass following total hip arthroplasty, making rice-like bodies cyst a valid one to consider. There is a wide array of possible differential diagnosis in front of an organized soft tissue mass following total hip arthroplasty . These iAn 82-year-old female patient presented to our clinics following progressive right hip nontender erythema, warmth, and swelling, with no functional impotence worsening progressively during the last month. She has no fever and no general weakness or loss of weight or any sign of infection or systemic disease.She has a past medical history significant for hypertension and well controlled diabetes with bilateral total hip arthroplasty for osteoarthritis. On the left asymptomatic side, she underwent a total hip arthroplasty at the age of 60 which was complicated, at age of 78, of periprosthetic infection and was treated with a two-stage revision of total hip arthroplasty. On the right symptomatic side, she underwent total hip arthroplasty at age of 67 which was complicated of nonunion of greater trochanter treated surgically with wiring at age of 74 and posterior dislocation at age of 77 that was treated surgically with a one-stage revision of the acetabulum due to instability. She was completely asymptomatic back then until the last month when she noted erythema, warmth, and swelling of the right hip. There was no history of rheumatoid arthritis, general systemic disease, or chronic infection conditions such as tuberculosis.Laboratory investigations were unremarkable with a normal WBC count, except for a CRP of 10 ng/mL and an ESR of 45 mm/hr. Radiographs of the pelvis showed bilateral noncemented total hip arthroplasty  with no significant soft tissue changes . No cystSurgical exploration was performed through an excision of the old lateral wound and by adopting the classic lateral approach of the hip. After cutting the gluteus medius, a large cystic mass formation was noted, overlying the hip joint and extending into it. This well contoured mass is opened revealing a collection of 250 mL of “rice-like” bodies measuring 1 cm each bathing in a clear fluid . It was All the taken cultures came back negative. The PPD was nonreactive. The RF and the anti-CCP serology were also negative. The microscopic study of the cystic collection and its content revealed a fibrotic synovial nature of cystic wall with fibrinous projections in the cystic cavity similar to the foreign objects qualified as “rice-like” bodies . In factThese formations are very rare and usually asymptomatic in the deep hip joint. However, they may cause pain by compressing nearby structures and groin edema when located anterolaterally , 3. InflMRI is considered as the imaging modality of choice for rice bodies which should appear as isointense or hypointense bodies in a magma of hyper-intense liquid on T2-weighted images –6. Some In front of a suffering patient, observation and conservative therapy are not an option especially that an active infection could not be ruled out. Percutaneous puncture and drainage which was indicated in this case according to the clinical and imaging findings failed . We opteThis is the second case of periprosthetic hip joint rice-like bodies cyst to be reported in literature . This evDespite many differentials that should be aroused in front of a slow growing symptomatic soft tissue mass complicating total hip replacement, a usually uncommon but actually more and more encountered possibility should be considered. This is the slow growing rice-like body cyst that may be found outside systemic conditions as a witness of a chronic local inflammatory condition. Therefore, as a result of our case report and of the one previously published, physicians should keep in mind this relatively uncommon condition when dealing with patients having a similar presentation.


Esophageal leiomyoma is the most common benign intramural tumor of esophagus. Although its incidence is not exactly known, it is very rare (0.006%-0.1% in autopsy series). It is generally asymptomatic and detected incidentally. Here, we present a rare case report describing coexistence of megaloblastic anemia and esophageal leiomyoma.How to cite this article: Coskun A, Unubol M, Yukselen O, Yukselen V, Aydin A, Şen S, Karaoglu AO. Esophageal Leiomyoma in Patients with Megaloblastic Anemia. Euroasian J Hepato-Gastroenterol 2014;4(2):98-100. If the treatment is delayed or failed, the symptoms would probably develop and it will be hard to differentiate it from malignancy.4 Here, we present the case of a coexistence of megaloblastic anemia and esophageal leiomyoma.Leiomyoma is the most prevalent benign tumor of esophagus. It is very rare, and the incidence in autopsy series is 0.006 to 0.1%. It is mostly observed in the mid 1/3 or lower parts of esophagus. Its characteristic feature is the proliferation of the smooth muscle layer, causing circumferential thickening localized on the esophagus wall. Frequently, it is observed as a single lesion. It can be hereditary or sporadic. Half of the patients are asymptomatic. Symptoms are generally associated with dysphagia, nonspecific retrosternal pain, heartburn and rarely weight loss. Esophagography, chest X-ray, computerized thorax tomography, thorax magnetic resonance imaging (MRI), esophagogastroduodenoscopy are useful for diagnosis. Recently, transesophageal ultrasonography has become widely preferred for preoperative diagnosis.12 vitamin level was 88 pg/ml , being highly low. In the bone marrow aspiration and biopsy examination, cellularity of the bone marrow was 80% and the ratio between myeloid series and erythroid series was 1:1. Megaloblastic changes were observed in erythroid series. B12 treatment was started for the patient who was diagnosed to have megaloblastic anemia due to vitamin B12 deficiency. With a view of megaloblastic anemia, esophagogastroduodenoscopy was done. In the lower 1/3rd of esophagus between 34th and 37th cm, a protrusion was found into the lumen and which was covered with normal mucosa, and partially with sub-mucosal vascularization. It was lobulated in part and probably intramural . He showed a HCT value of 19.1%, MCV of 111.7 fl, RDW of 26.1%, leukocyte count of 2,900/ml, neutrophil count of 55%, lymphocyte count of 37%, monocyte count of 5%, eosinophil count of 3%, thrombocyte count of 123.000/ul In the peripheral blood smear of the patient who was diagnosed as pancytopenia, there were macrocytosis, anisocytosis in erythrocytes and hypersegmentation in neutrophils. Reticulocyte count was 1%. Serum Btramural ./ml and thrombocyte 266.000/ml. The lesion was observed to have the same properties as before. The patient reported that he had developed no complaints in the meantime. Endosonography was performed. It was found as a mass at distal esophagus, starting from incisor teeth, in between 34th and 37th cm (37.5 × 19.5 mm in size), submucosally located, having smooth contours, heterogeneous hypoechoic and interpreted as compatible with mesenchymal tumor , contrast involvement and circular wall thickening was observed in a segment of 4 cm at the distal end of esophagus. No extension toward surrounding soft tissues was found. In the biopsy sample, neoplastic cells were detected with actin and desmine, while negative with vimentin. Mitosis was not determined at sections, pathology result was determined as leiomyoma .3 Esophageal leiomyoma constitutes 10% of all gastrointestinal system leiomyomas.1 Characteristic feature of leiomyoma is the proliferation of smooth muscle layer that causes circumferential thickening localized at esophagus wall. It is well-circumscribed, surrounded by a capsule of fibrous tissue, sessile, benign and spiral-shaped/whorled tumor. Tumor lesions are generally small, solitary with excentric position, solid, round and easily removable from the capsule. Generally, it is diagnosed between 20 and 50 years of age. It is observed more than twice in men than women.5 Our patient was a 52-year-old man. In 3 to 10% of cases, multiple numbers of lesions occur.5 Leiomyomas are usually intramural. However sometimes, these can be detected in the proximity of or inside of esophageal diverticulum.6 They can undergo cystic degeneration; however, progression to malignancy is rare. About 800 cases have been identified in the literature. Only in two of them, malignant transformation into leiomyosarcoma was observed (0.2%).7 In our case, the tumor lesion showed no change within 4 years. Nevertheless, leiomyomas accompanying/coexisting with malignant neoplasms have been defined in literature. It can be hereditary or sporadic. It has been related with Alport syndrome among hereditary diseases.5 If tumor exceeds mucosa and ulcer develops, bleeding may occur. In symptomatic patients, tumor diameter is observed (average 5.3 cm). In asymptomatic patients, its average diameter is about 1.5 cm.1 Generally, it grows slowly. The use of endoscopic ultrasonography in the diagnosis of esophageal leiomyoma is increasing.3 It is a considerably reliable method in differentiation of solid and cystic submucosal esophageal masses as well as in viewing these lesions. This method can be used in localizing mediastinal lesions and mural lesions of esophagus. In treatment, even if the leiomyoma is asymptomatic, it is advised to operate. Otherwise, malignancy cannot be eliminated. In differentiating from malignity, definite diagnosis must be done by excision and histological examination. Enucleation is a reliable and effective method of treatment. It provides relief in all symptomatic patients. Perioperative morbidity and mortality is not observed.1 In its monitoring, malignant transformation and recurrence tendency has not been observed. Intra-operative esophagoscopy combined with video-assisted thoracoscopic approach is the method used for easing the process and shortening the length of hospitalization. However, removal of all small asymptomatic leiomyoma may be unnecessary. In megaloblastic anemia, various idiopathic cases, such as gastric cancer and carcinoid, can be observed in gastrointestinal system.89 A single endoscopy should be considered to identify prevalent lesions  in patients with pernicious anemia, but there are insufficient data to support routine subsequent endoscopic surveillance for these patients.10 In case of megaloblastic anemia if gastrointestinal symptoms are seen, upper endoscopic examination may be useful.Less than 10% of esophagus tumors are benign tumors and 4% of them are comprised of leiomyoma.


Vena cava thrombus is an extremely rare complication of testicular tumors. We report on an unusual case of testicular tumor presenting with inferior vena cava thrombus extending from the left spermatic and bilateral external iliac veins to the hepatic vein.A-35-year old man presented with a 6-month history of left scrotal mass and a 1-day history of bilateral lower extremity edema. Computed tomography (CT) revealed the presence of thrombus extending from the left spermatic vein and bilateral external iliac veins to the hepatic vein, and multiple lymph node and lung metastases. 3 cycles of chemotherapy were given after the left high inguinal orchiectomy. Pathological examination demonstrated a pure yolk sac carcinoma with lymphovascular invasion and direct tumor extension into the left spermatic cord. CT and positron emission tompgraphy-CT obtained no findings of metastasis or recurrence at 3 months after the chemotherapy.We review this seldom case and discuss the literature with regard to its diagnosis and treatment. Inferior vena cava thrombus due to a testicular tumor is an extremely rare condition. There have been a few reports of thrombus due to testicular tumor extended to inferior vena cava in literature. Most of pathological examinations revealed mixed germ cell carcinoma which generally consisted of embryonic carcinoma –3. We prA 35-year-old man was admitted to the hospital complaining of bilateral lower extremity edema. He reported a six-month history of painless mass in the left scrotum. Physical examination disclosed a 10 × 6cm firm non-tender left scrotal mass. A thorax computed tomography (CT) demonstrated multiple metastases in both lungs. Abdomen enhanced CT revealed retroperitoneal multiple lymph node metastases, inferior vena cava thrombus extended from the left spermatic vein and bilateral external iliac veins to the hepatic vein and a decrease in the function of the left kidney due to a thrombus of the left renal vein . Color DA left high inguinal orchiectomy was performed and pathological diagnosis was pure yolk sac carcinoma with lymphovascular invasion and direct tumor extension into the left spermatic cord. Three cycles of bleomycin, etoposide, and cisplatin (BEP) were given. A nearly complete response in the retroperitoneal and lung metastases was observed by thorax and abdominal CT , tumor mThrombus of inferior vena cava due to testis tumor is an extremely seldom condition. In earlier cases –7, most Since the inferior vena cava thrombus lengths of earlier cases had been generally short, these patients were treated with either chemotherapy alone or surgery excision of thrombus after the chemotherapy –7. Only Our case had inferior vena cava thrombus extended from the left spermatic vein and bilateral external iliac veins to the hepatic vein. The radiological imaging (CT and PET-CT) indicated no evidence of metastasis or recurrence after orchiectomy and chemotherapy. Although the inferior vena cava remained occluded completely, there were evidences of collateralization such as the improvement of the left renal function and disappearance of lower extremity edema. Overall, we discussed with the present patient and decided not to performed surgery excision of inferior vena cava thrombus.Testicular tumor presenting with thrombosis of the inferior vena cava, with or without subsequent pulmonary embolism, can be managed with a combination of anticoagulation and/ or inferior vena cava filter placement. The present patient was begun anticoagulation therapy before the chemotherapy for the risk of pulmonary embolism but a filter was not placed into the inferior vena cava. After partial pulmonary embolism occurred, the filter was placed into inferior vena cava. We suggest that a filter should be placed and started anticoagulation therapy before the chemotherapy because of the risk of pulmonary embolism.


Diagnosis and preoperative planning are critical in the execution of any surgical procedure. Panoramic radiography is a routine method used in dentistry to assist clinical diagnosis; however, with this technique 3D anatomical structures are compressed into 2D images, resulting in overlapping of structures which are of interest in the diagnosis. In this study we report the case of a patient who presented with a dentigerous cyst of expressive dimensions in the body of the mandible region. The surgery was planned and executed after observing the margins of the lesion by Cone Beam Computed Tomography (CBCT). We conclude that CBCT is a precise method to help diagnosis; it provides greater accuracy in surgical treatment planning through 3D image display, allowing more effective results.  The dentigerous (or follicular) cyst (DC) is the second most common type of dental cyst and the most common in jaw development . Benn anDiagnosis of the lesion should not be based on X-ray evidence alone, but also on clinical evidence, particularly microscopic examination of the sample .Periapical and panoramic X-rays are the most commonly used imaging examinations in dentistry for diagnosis and surgical planning. However the information acquired in these examinations is limited, since the three-dimensional anatomy of the area X-rayed is shown in two dimensions, with superimposed planes. Although these methods produce acceptable images in the mesiodistal direction, observation in the vestibulolingual direction is difficult. There may also be geometrical distortion of the structures X-rayed when we use these imaging methods . A very In the last decade a new technology—Cone Beam Computerised Tomography (CBCT)—has offered dental surgeons three-dimensional reproduction of images of mineralised maxillofacial tissues, with minimum distortion and significantly lower doses of radiation than conventional CT .The object of this study was to report a clinical case of a dentigerous cyst in which the diagnostic hypothesis and the decision on the treatment plan were based on CBCT.Male patient, 28 years, Brazilian, white, attended a private dental practice complaining of increased volume in the vestibular region of the left mandibular second molar and slight discomfort when eating. The patient reported that the increase had evolved slowly over approximately one year, with no apparent cause and no pain. In clinical examination, the absence of the left mandibular second molar was observed, with an increase in volume in the vestibular region.CBCT was carried out to evaluate the case. The CBCT images were obtained with i-CAT® Next Generation, 120 Kv, 18.5 mA, FOV 16 × 6 cm. The data were converted into 3D images with volume representation by the Vision Standalone software. In the axial sections an extensive, hypodense image was observed in the body of the mandible region, left side, containing the permanent mandibular second molar; bulging of both osseous cortices was also found . In the The diagnostic hypothesis based on the clinical and imaging examinations was of a dentigerous cyst of the circumferential radiographic type. Because the lesion presented with relatively unaggressive comportment, with large size and limits, and was located very close to important anatomical structures, we decided on conservative surgical treatment, that is, decompression of the cyst and subsequent enucleation.The procedure practised on the patient started with aspiration puncture, which confirmed the presence of an intralesional liquid content. After confirmation of a cyst-type lesion, an incision was made to give access to the cyst capsule and to promote biopsy. The surgical piece obtained was sent for histopathological examination, resulting in the histological diagnosis of a dentigerous cyst.Dentigerous cysts account for 30.7% of all odontogenic cysts in the Brazilian population , being cDentigerous cysts are usually located in the region of the maxillary canines and the mandibular third molars , 12, 13;Hyomoto et al.  analysedCBCT has been widely used in dentistry to provide basic information on dental and maxillofacial structures for diagnosis and surgical planning . AccordiDentigerous cysts are usually discovered during routine X-ray examinations or when X-ray is indicated to determine the reason for a failed tooth eruption . PanoramImaging is an essential part of preparation for most surgical procedures . PreoperIn general, surgery is recommended for dentigerous cysts because they often block tooth eruption, become large, displace teeth, destroy bone, and invade vital structures; in some cases they may cause fractures , 26. In According to Neville et al. , the levIn conclusion we may say that CBCT is a very useful complementary tool for diagnosis and surgery planning in cases of dentigerous cyst. Three-dimensional viewing of the structures using this imaging method offers greater accuracy in planning surgical treatment, thus allowing more effective results to be achieved.


FNAC) is of limited use. Radiological evidence demonstrating displacement of blood vessels may aid diagnosis of schwannoma. Malignant transformation of ancient schwannoma has been reported. Surgical excision carries a high risk of postoperative Horner's syndrome.Ancient schwannomas exhibit characteristic histological features. Fine‐needle aspiration cytology ( Schwannomas are typically benign, slow‐growing, solitary neoplasms of Schwann cell origin. They were first described by Verocay in 1908 A 36‐year‐old female, nonsmoker, presented to the head and neck rapid access clinic with a 1‐month history of a painless, left‐sided, neck lump. There was no preceding illness or features in the history suggestive of malignancy. Examination confirmed the presence of a soft, mobile left level II neck lump measuring approximately 2.5 cm in diameter. There were no overlying skin changes. Flexible nasendoscopy and examination or the oropharynx was normal.Ultrasonography showed an isolated pathological left level II node measuring 1.4 cm in the short axis but no other pathological nodes. FNAC was nondiagnostic, producing hemorrhagic aspirate with inadequate cellular yield. Due to the proximity of the lymph node to the carotid sheath, magnetic resonance imaging (MRI) scan was chosen in preference to ultrasound‐guided Trucut biopsy  yields nondiagnostic hemorrhagic aspirate and is of limited use.Radiological evidence of displacement of blood vessels can aid with the differential diagnoses of schwannomasMalignant transformation in an ancient schwannoma has been reported.Careful dissection of the mass from the nerve sheath is required to reduce the risk of postoperative Horner's Syndrome.PFB: design, literature review, and quality assurance of manuscript. RCTC: design, literature review, write‐up and quality assurance of manuscript. SC: design, literature review and quality assurance of manuscript. AP: design, quality assurance and principal surgeon on clinical case.The authors declare that there is no conflict of interest regarding the publication of this paper.


Treatment success of periodontal-endodontic lesions is dependent on the elimination of both disease causative factors, whether they exist separately or concurrently. This report presents successful endodontic management of a misdiagnosed large periradicular pathology, which had not resolved after a previous periodontal regenerative surgery. A patient complaining of discomfort in the left maxillary region was referred. He had undergone regenerative surgery for treatment of a large periradicular defect; however, there was no further amelioration of the clinical signs/symptoms. Radiographically, a large periradicular lesion filled with bone substitute materials was detected around tooth #25. The endodontic treatment of the tooth was imperfect; therefore, surgical endodontic retreatment was planned. During root-end surgery, the biopsy containing bone substitute materials was obtained. Root-end filling/sealing using calcium-enriched mixture cement was completed. The histopathological examination showed granulation tissues enclosing exogenous materials. In two-year radiographic evaluation, resolving lesion and complete bone healing was observed. The first fundamental step in the management of periradicular lesions is correct diagnosis of the lesion origin and set-by step of the treatment plan according to the main causative factor. Regenerative periodonttal surgery, without considering the defective apical seal, will only cause a painful procedure for the patient without any positive benefit. Following appropriate apical seal, the endodontic lesion healing can be anticipated.  In differential diagnoses of periradicular lesions, the lesions with primary endodontic origin have the major contribution. However, the clinicians should also consider lesions of non-endodontic origin, including anatomic variations, other odontogenic or developmental cysts or neoplasms, and different categories of combined periodontal-endodontic lesions. Each one needs different treatment plan and has different prognosis , 2.The pulp and periodontium are anatomically and functionally interrelated from the embryonic period to all over the life, in health and disease . EndodonIn primary endodontic lesions, resorption of the adjacent periapical bone and destruction of the attachment apparatus would happen. The suppurative process may establish a sinus tract that can extend through the periodontal ligament space and apical foramen. In such lesions with secondary periodontal involvement, consequent to a non-healed endodontic lesion, and as the result of continuing drainage and massing of plaque and calculus in the pocket, the periradicular alveolar bone would destroy further and can proceed into more apical migration of the attachment and establishing periodontal disease .While a radiolucent lesion surrounds a previously root-treated tooth, the diagnosis and the treatment plan can be more complicated [6]. In the cases of failed conventional root canal treatment, nonsurgical retreatment can be the first choice. When nonsurgical retreatment is not practical or is predicted to have low success rate, the treatment plan would be surgical endodontics . In thisThis report presents successful management of a misdiagnosed large periradicular pathology. First, a periodontist intended to treat this lesion by a regenerative surgical procedure. As the operation outcome was not successful, the patient sought for treatment again.A 30-year old man with frequent pain, discomfort and swelling in the left maxillary premolar area was referred to a private clinic. The patient stated that he had sought treatment for the problem by visiting a periodontist 6 months before. The specialist had performed a regenerative periodontal surgery in the region which resulted in no symptom relief. In clinical evaluation, the tooth #24 showed no caries and had normal response to pulpal sensibility tests. It had normal probing depth and was not sensitive to percussion. Tooth #25 had a metal-ceramic crown. It was obviously painful on percussion and the adjacent vestibule was so sensitive to palpation. The gingival mucosa showed slight swelling and redness. He had also complaint about chewing with the tooth. Probing examination showed normal attachment and depth (≤3mm) and no mobility was observed. In radiographic assessments , a largeBecause of the surgical history, poor quality of the root canal therapy and existence of a casting post and core, the treatment plan decided to be a surgical endodontic retreatment. It was discussed with the patient and an informed consent was obtained. An endodontist carried out the surgery. Following local anesthesia with 2% lidocaine with 1:80000 epinephrine , a full mucoperiosteal flap was raised. After flap reflection, the lesion was partially curetted to remove the inflamed tissues containing bone substitute  and gainAfter root-end resection, the root-end cavity was prepared with an ultrasonic retrotip .Calcium-enriched mixture (CEM) powder and liquid  were mixed according to the manufacturer’s instructions. The biomaterial was inserted into the prepared cavity to achieve root-end filling/sealing. Subsequent to taking a confirmation radiography , the flaThe patient was recalled 7 days later. Clinical assessments showed absence of signs/symptoms, and the patient did not have any complaint about chewing with the tooth. On 6-month follow-up, the tooth was asymptomatic and functional. Healing of the lesion was uneventfully in progression . On two-This case report describes management of a large periradicular periodontitis, first misdiagnosed and mismanaged. Correct diagnosis is the crucial prerequisite for determining treatment strategies and long-term prognosis . On someNevertheless, the primary endodontic lesions often cure following disinfection and sealing the root canal system; and one-year follow-up radiographic examinations usually demonstrates bone healing in the area . Thus, iThe reported case was about a periradicular inflammatory odontogenic lesion with endodontic origin. There is similarity in clinical and radiographic aspects of primary endodontic disease with secondary periodontal involvement, primary periodontal disease with secondary endodontic involvement and true combined pathologies . In spitWhat initiates periodontal disease is the microbial dental plaque and clinical investigations present the improvement in periodontitis resulting from improved plaque control; thus, accumulation of plaque and calculus and poor oral hygiene are the main environmental causative factors for periodontitis , 21. TheIt can be assumed that maybe the previous insertion of bone substitute materials  in the presented case might provide the matrix for the new bone regeneration in the region after elimination of the etiologic factors. Non autogenous bone replacement grafts can provide significant clinical improvements in osseous defects in comparison to surgical debridement alone . In someAn ideal root-end filling biomaterial should be able to create a three-dimensional seal and promote cementogenesis , 30. ManTo treat a large odontogenic lesion, the first important step is precise diagnosis of the lesion aetiology. Regenerative periodontal surgery, without considering the imperfect endodontic seal, might lead to treatment failure. Following appropriate apical seal using new root-end filling biomaterials, healing of the lesion can be predicted.


Salivary gland choristoma of the middle ear cavity is a very rare condition. These lesions are a result of a defective embryonic development and their adjacent structures may be associated with abnormalities. Here we report a case of salivary gland choristoma of the middle ear who presented to Be’sat Hospital, Tehran, Iran in 2015 with unilateral conductive hearing loss. There are 41 case reports in English and non-English literature from 1961. Taylor and Martin reported the first case of middle ear salivary choristoma. Choristoma is not a tumor, but it is a histologically normal mature tissue located in an ectopic position . The preSalivary gland choristoma of the middle ear cavity was first reported by Taylor and Martin in 1961, and since then only 41 cases have been reported . The mosHere, a typical case of salivary gland choristoma of the middle ear is described who presented with conductive hearing loss in his left ear developed during the patient’s childhood.A 39-yr-old man with a hearing loss that had persisted since childhood in his left ear and an intermittent otorrhea and otalgia was presented to Be’sat Hospital, Tehran, Iran in 2015. The previous episode of otorrhea and otalgia from childhood treated with antibiotics findings from his physical examination showed no external deformities of his auricle and external ear canal. Otoscopic examination revealed opacity at the lower part of his left tympanic membrane. Pure tone audiometry results were normal in his right ear, but in the left ear demonstrated 56-db conductive hearing loss with normal bone conduction . The masExploratory tympanotomy of the left ear was performed under general anesthesia. A 1 0.7 cm yellowish mass adherent to the mucosa of the promontorium was found. No evidence of cholesteatoma was observed. The malleus and the incus were intact, but the stapes had been eroded by the mass. The mass was easily dissected. The respiratory epithelium covering seromucinous glands were found in histopathologic examination of the removed mass . Pure toThe middle ear salivary gland choristoma is a rare condition. About 41 cases (including the present case) have been reported in the literature , 7. AlthThis lesion may accompany by other branchial arch anomalies . The disOur patient was a 39-yr-old man with clinical features including unilaterality and left sided occurrence. This is in agreement with other studies , 11. HeaMost injuries associated with middle ear salivary gland choristoma are due to the sole presence and action of the choristomatous mass and not resulting from embryological failure or abnormal development of the first and second branchial arches . Facial In our case, involvement of the facial nerve or abnormalities in the course of the facial nerve was not observed. Any histopathological investigation can establish the definite diagnosis of salivary gland choristoma , 6. ThisTreatment is determined by the size and location of the tumor . ConservSalivary gland choristoma is an unusual condition and. It should be diagnosed and differentiated from other lesions of the middle ear that lead to conductive hearing loss. However, in pedunculated and small tumors, total excision has been recommended without causing permanent damage to the facial nerve, because several cases with malignant neoplasm arisen from choristoma have been reported.The authors declare that there is no Conflict of Interests.


Bouveret’s syndrome, first described in 1896, is an unusual cause of gastric outlet obstruction secondary to large gallstone impaction in the proximal duodenum after migration through a cholecystoduodenal fistula. Stone migration has been previously described after endoscopic or surgical fragmentation. However, this is the first reported case, in our knowledge, where the stone migrated after oral contrast administration a few days after the onset of symptoms, causing a distal gallstone ileus. Bouveret’s syndrome, first described in 1896, is an unusual cause of gastric outlet obstruction secondary to large gallstone impaction in the proximal duodenum after migration through a cholecystoduodenal fistula. Stone migration to the distal small intestine has been described after primary endoscopic or surgical fragmentation of the original impacted gallstone [A 61-year old female with diabetes mellitus and hypertension presented with a five-day history of epigastric pain and postprandial vomiting. A physical examination revealed a soft abdomen with mild epigastric tenderness. Labs were significant for an acute kidney injury. An abdominal CT scan following water-soluble hyperosmolar radiocontrast administration showed a 3.5 cm intraluminal calculus at the level of the third duodenal segment, diffuse pneumobilia, and an air-containing gallbladder, suggesting Bouveret’s syndrome (Figure An initial improvement was noted later, followed by the recurrence of symptoms two days later. A repeat abdominal CT scan showed the migration of the duodenal stone and impaction at the distal jejunum with the dilation of the proximal loops, suggesting gallstone ileus Figure .Subsequent jejunotomy and the successful extraction of the stone Figure  were perAlthough over 300 cases of Bouveret’s syndrome have been reported, it is unusual to see a migrating gallstone after seven days of obstruction. The rarity of stone migration and its timing after the ingestion of the water-soluble contrast raised the speculation of possible “iatrogenic” migration of the stone. Conventional hyperosmolar water-soluble contrast media have pharmacologic effects, including cholinesterase inhibition, the release of histamine, and in the gut, the release of a serotonin-like substance, which enhances bowel motility -4.We believe that the hyperosmolar contrast agent played a role in inducing osmotic pressure, intestinal smooth muscle contraction, and decreasing wall edema, which led to the spontaneous disimpaction and migration of the stone forwards. We believe this presentation is unique and warrants a heightened clinical awareness in order to avoid delayed diagnosis and management.


Intraventricular cavernous angiomas are rare pathological entities, and those located at the foramen of Monro are even rarer. We herein present a case of cavernous angioma at the foramen of Monro that was successfully treated by neuroendoscope-assisted surgical removal, and review the relevant literature.A 65-year-old woman had experienced headache and vomiting for 10 days before admission to another hospital. Magnetic resonance imaging (MRI) showed a mass at the foramen of Monro, and obstructive hydrocephalus of both lateral ventricles. The patient was then referred to our hospital. Neurological examination on admission to our hospital showed memory disturbance  and wide-based gait. A cavernous angioma at the foramen of Monro was diagnosed based on the typical popcorn-like appearance of the lesion on MRI. The lesion was completely removed by neuroendoscope-assisted transcortical surgery with the Viewsite Brain Access System , leading to a reduction in the size of the ventricles. The resected mass was histologically confirmed to be cavernous angioma. The patient’s symptoms resolved immediately and there were no postoperative complications.Minimally invasive neuroendoscope-assisted surgery was used to successfully treat a cavernous angioma at the foramen of Monro. Intraventricular cavernous angiomas are rare pathological entities, constituting 2.5–10.8 % of cerebral cavernous angiomas  showed enlargement of both lateral ventricles and a mass at the foramen of Monro. The patient was then referred to our hospital.Neurological examination on admission to our hospital showed memory disturbance  and a wide-based gait disturbance. Computed tomography (CT) showed a 16 mm mildly hyperdense mass with no calcification at the foramen of Monro, and the mass was causing obstructive hydrocephalus. MRI revealed a well-delineated mass at the foramen of Monro with heterogeneous signal intensity on both T1- and T2-weighted images; the mixed-signal core appeared as a popcorn-like lesion typical of cavernous angioma , endoscope-assisted surgery in 2 patients , and endoscopic surgery 2 patients .Thirteen patients demonstrated full recovery of neurological function after surgery. Of the other four patients, one retained mild neurological deficits due to postoperative hydrocephalus, one fell into a persistent vegetative state due to massive hemorrhage at symptom onset, one who was found unresponsive with decorticate posturing to noxious stimuli died, and one was not available for follow-up.The treatment goal in patients with cavernous angioma is total removal because postoperative remnants increase the risk of regrowth and bleeding. Intraventricular tumors and cysts are ideal lesions for the application of neuroendoscopy; good visualization is possible because of their location inside the CSF-filled ventricular system, and the often-associated obstruction of the CSF pathway and ventricular enlargement offer the possibility of working in large spaces , associated hydrocephalus, and low histological grade . In the present case, we chose endoscope-assisted resection because we could use the Viewsite as an access port to enable dual instrumentation  and work in an air ambience. We performed bimanual dissection by freeing both hands through a smaller skin incision, craniotomy, and corticotomy and inducing minimal white matter damage  and allows dynamic magnification  is a tubular retractor system designed specifically for intracranial use. It consists of an introducer that permits entry into the tissue and a working channel, and it has transparent plastic walls that permit visualization of surrounding tissue (Raza et al. The use of neuronavigation in preoperative trajectory planning and establishment of intraoperative landmarks to avoid morbidity was helpful when the lesion overlapped the fornix and feeding vessels of the choroid plexus (Prat and Galeano We experienced an extremely rare case of cavernous angioma at the foramen of Monro. Minimally invasive neuroendoscope-assisted surgery was used to successfully remove the tumor without complications.


This presentation of two cases and literature review discusses the epidemiology, clinical manifestations, pathogenesis, diagnosis, treatment, and prognosis of high-grade glioma with extracranial metastases.A retrospective analysis of the clinical features of two cases of malignant glioma, including metastatic sites, pathological data, and treatment methods, and a literature review was performed.Two patients developed extracranial metastases within 1 year after surgery for primary glioma. One patient developed cervical lymph node and bone metastases while the other developed bone metastases, and both patients died within 2 months after the diagnosis of the extracranial metastasis.Extracranial metastases may develop from malignant gliomas. According to the literature, the most common extracranial site is intraspinal , followed by the vertebrae, lungs, liver, and lymph nodes. The complex metastatic mechanism remains unclear, and the prognosis is very poor, with a survival duration of less than 6 months. Malignant glioma is a devastating neurological disease with a uniformly poor prognosis and a clinical course characterized by progressive functional and cognitive impairments . ExtracrHigh-grade gliomas are particularly insensitive to radiation and genotoxic drugs . Unlike Surgery is the first-line therapy for glioma. However, gliomas recur in the resection margins of 90% of patients . Many stThe vast majority of glioma recurrences occur within 2 cm of the primary tumor area ; accordiA 43-year-old man was hospitalized after complaining of intermittent headache and dizziness that progressed in severity for 1 month, as well as an unsteady gait for 2 weeks. Brain magnetic resonance imaging (MRI) revealed high T1 and T2 signals in the left occipital lobe (4 × 3 cm) with peripheral edema and lateral ventricle compression, and a pathological analysis after tumor resection indicated glioblastoma. Although the patient was treated with conformal radiotherapy, metastases of glioblastoma were found in the chest and back, as well as in the left neck Fig.  depicts A 41-year-old woman who had undergone tumorectomy 9 months earlier was hospitalized after complaining of pain in the lumbosacral region for 2 weeks. The patient had previously been diagnosed with glioma  stage III) in the right temporal lobe and was treated with radiotherapy  and intravenous chemotherapy . She had no brain neurological symptoms. Positron emission tomography (PET)-CT revealed a sacral metastasis; however, local radiotherapy  provided no pain relief. The patient refused to continue radiotherapy and died 1 month after the detection of the metastatic lesions. We note that both patients had previously been healthy, with no history of other diseases.In this report, we describe two patients who developed extracranial metastases after 1 year: one developed cervical lymph node and bone metastases, and one developed a bone metastasis. Both patients died within 2 months after the extracranial metastases were diagnosed.Neurosurgeons and oncologists widely believe that malignant gliomas never metastasize outside the central nervous system (CNS). However, this notion has been gradually proven incorrect . ExtracrIn the two above-described cases, the surgery for the primary lesion and the diagnosis of the metastases were separated by intervals of 3–9 months, and the lesions were located in the left neck, chest, and sacral bones. According to the literature, the average age of patients at the time of malignant glioma diagnosis was 38–42.2 years , the medLymph node, vertebral, and sacral metastases were confirmed in both of our patients. The histological types were glioblastoma multiforme (case 1) and anaplastic astrocytoma (case 2). The most common site of extracranial metastasis, according to previous reports, is intraspinal  , followeAlthough the extracranial metastatic mechanism is currently unclear, iatrogenic spread consequent to surgery may be the main cause of extracranial metastases , 26. TheClinical manifestations are based on the sites of extracranial metastases, and symptoms are usually related to the affected organs and tissues. Patients with symptoms and signs in corresponding sites should be subjected to evaluation with an appropriate ancillary modality, such as CT, MRI, single-photon emission CT (SPECT), or PET-CT. MRI is preferred for the evaluation of cases involving symptoms caused by spinal cord or nerve root lesions, MRI and SPECT can be used to evaluate bone pain, and PET-CT may be selected for some cases. If biopsy of the metastases is feasible, tissues from the lesions should be obtained and subjected to pathological examination .A system of diagnostic criteria for extracranial metastases of malignant glioma has been developed. Weiss  proposedCurrently, no standardized protocol exists for the treatment of extracranial metastasis, which is palliative. Most patients receive radiotherapy and/or chemotherapy, and no significant difference in clinical efficacy has been observed between these treatment methods , 27, 28.As noted, patients with extracranial metastases of malignant glioma have a very poor prognosis and a life expectancy of less than 6 months , 33. TheAlthough surgery is the preferred treatment strategy for malignant glioma, it can damage the blood-brain barrier, thus allowing tumor cells to form extracranial metastases. Therefore, surgeons should carefully ensure the principle of a non-tumor operation and maintain an intact dura. As described in the literature , adherenIn summary, the glioblastoma might undergo extracranial metastasis. Although we have described extracranial metastases of malignant gliomas in detail, the complex mechanism underlying metastasis remains unclear. Since the metastasis is not sensitive to chemoradiotherapy, the operation should strictly adhere to the tumor-free principle and try to guarantee completeness of cerebral dura mater for the sake of reducing the occurrence rate of extracranial metastasis. Technological advances in the diagnosis and treatment of malignant gliomas and gradual increases in patient survival may lead to a gradual increase in the incidence of extracranial metastases in the future. Currently, however, patients with extracranial metastases of malignant glioma have a poor prognosis, and most die within 6 months of diagnosis.


Fistulae between the colon and upper gastrointestinal tract are distressing and uncommon complications of malignancies involving this region. We report a case of a middle-aged man with a locally advanced and metastatic distal transverse colon malignancy who presented with a duodenocolic fistula proximal to the primary tumor and underwent palliative surgery.A 50-year-old Sri Lankan man presented to our hospital with a history of feculent vomiting of 1 week’s duration preceded by worsening constipation and abdominal fullness of 2 months’ duration. He also complained of anorexia and significant weight loss over the previous month. His physical examination was unremarkable except for his wasted appearance. Flexible sigmoidoscopy done at his local hospital had not revealed any abnormality in the left colon. Gastroduodenoscopy did not reveal fecal matter or any mucosal abnormalities in the stomach or duodenum. An abdominal contrast-enhanced computed tomographic scan showed a mid-to-distal transverse colonic tumor with a duodenocolic fistula proximal to the primary lesion. At laparotomy, he was found to have an unresectable, locally advanced mid transverse colon tumor with diffuse peritoneal and mesenteric deposits and mild ascites. Palliative end ileostomy and gastrojejunostomy were performed before closure. Histology from the malignant deposits revealed a well-differentiated adenocarcinoma. He made an uneventful recovery with good symptomatic relief.Malignant gastric or duodenocolic fistulae are uncommon complications of locally advanced colonic malignancies with direct invasion to the stomach or duodenum. Although the characteristic clinical presentation of feculent vomiting suggests the diagnosis, cross-sectional imaging is confirmative in addition to staging the disease. Management is guided by disease stage, nutritional status, and the general condition of the patient and ranges from extensive bowel resection including the fistula to palliative options. Enteroenteric fistulae complicate malignancies and benign conditions such as Crohn’s disease, tuberculosis, actinomycosis, radiation enteritis, diverticulitis, and peptic ulcer disease, and they may rarely be iatrogenic –4. Malig2. His general and abdominal examinations were otherwise unremarkable.A previously healthy 50-year-old Sri Lankan man presented to our hospital with feculent vomiting of 1 week’s duration. This was preceded by worsening constipation, abdominal fullness, and intermittent abdominal pain of 2 months’ duration. He denied passage of fresh or altered blood with stools. Interestingly, the abdominal discomfort and fullness and the sensation of a full rectum were at times relieved by feculent belching. He had significant weight loss over the previous month and associated anorexia. He was a nonsmoker and had not been on nonsteroidal anti-inflammatory drug therapy. He was dehydrated and appeared wasted, with a body mass index of 21 kg/mHe had been investigated at his local hospital, where a flexible sigmoidoscopy had not revealed any abnormality in the left colon. Suspecting a malignant gastrocolic fistula, a gastroduodenoscopy was performed, but this did not reveal fecal matter or any mucosal abnormalities in the stomach or duodenum. An abdominal contrast-enhanced computed tomographic (CT) scan revealed a mid-to-distal transverse colonic tumor with a duodenocolic fistula proximal to the primary lesion Fig.  and b. AAt laparotomy, he was found to have an unresectable locally advanced mid-to-distal transverse colonic tumor with possible infiltration of the duodenojejunal (DJ) flexure. A small amount of ascites, diffuse peritoneal and mesenteric deposits with scirrhous contraction of the mesocolon and small bowel mesentery, was noted Fig.  and b. APostoperatively, he received eight cycles of FOLFOX therapy , for which he showed an excellent response and was well and disease-free 10 months after surgery.Duodenocolic fistulae are an uncommon form of gastrointestinal fistulae and a rare presentation of colonic malignancies around the hepatic flexure , 2. As iCross-sectional imaging is the cornerstone of diagnosis and defines the anatomy of a suspected gastro- or duodenocolic fistula. Differentiating a duodenocolic fistula from a gastrocolic fistula is important for planning surgery. Abdominal contrast-enhanced CT scans play a vital role in delineating the fistula while also assessing locoregional spread and metastatic disease.Endoscopy, though performed in this patient, failed to demonstrate either the tumor or the fistula. Failure to identify the tumor early was due to its laying beyond the reach of the sigmoidoscope, and a colonoscopy was warranted. Failure to identify the fistula by gastroduodenoscopy may have been the result of an incomplete or inadequate study, or it may have been due to the fact that the tract opened up to discharge colonic contents only when there was a rise in intracolonic pressure. An oral contrast study, if done preoperatively, may not have demonstrated the fistula for the same reason.Treatment of malignant duodenocolic fistulae depends on several factors, such as the extent of local invasion, distant metastasis, and the general condition and comorbidities of the patient. Preoperative correction of fluid and metabolic abnormalities is essential for improved outcomes . PatientSurgery is either curative or palliative, depending on the stage of the disease. Examples of curative strategies are a right colonic resection including the fistula tract and duodenal wall with primary closure of the defect or the use of a jejunal drainage loop or patch , 11. AltThe metastatic and locally advanced disease in our patient necessitated a palliative approach. Palliative surgery is indicated when there is extensive local infiltration with retroperitoneal extension, extension to the major vessels, and distant metastasis. An ileotransverse anastomosis combined with a gastrojejunostomy may be performed in selected patients with advanced disease to relieve the obstruction and defunction the fistula . BypassiMalignant gastric or duodenocolic fistulae are uncommon complications of locally advanced colonic malignancies with direct invasion to the duodenum. Although the characteristic clinical presentation of feculent vomiting suggests the diagnosis, cross-sectional imaging is confirmative in addition to staging the disease. Management is guided by disease stage, nutritional status, and the general condition of the patient and ranges from extensive bowel resection including the fistula and palliative options. Adjuvant chemotherapy may be of benefit in selected patients following surgery.


A tumefactive lesion of central nervous system (CNS) is defined as a mass-like lesion with a size greater than 2 cm in brain detected by magnetic resonance imaging (MRI). Neuroimaging may help to distinguish the nature of a tumefactive lesion and therefore, can prevent an unnecessary brain biopsy. Here we emphasized on determining the nature of a CNS tumefactive lesions with the help of MRI and more explanations about demyelinating lesions with focus on Schilder and Balo diseases as two multiple sclerosis variants. We have reported here two boys of 10 and 8 years of age respectively of multiple sclerosis (MS) variants who presented with acute neurologic complications to our hospital as one of the two referral children hospital in Tehran, Iran. Tumefactive demyelinating lesions can be considered a separate entity that itself can contain Schilder disease, Balo disease, some cases of acute disseminated encephalomyelitis (ADEM) or classic MS. MRI can help to establish a diagnosis of a tumefactive lesion and to differentiate among different underlying etiologies.  Magnetic resonance imaging (MRI) of the central nervous system (CNS) is essential for initial investigation of a child presented with acute or sub-acute focal neurologic problems, which may show a mass-like lesion. The four differential diagnoses of a tumefactive (mass-like) CNS lesion found on MRI are tumors and infiltrative lesions, vascular lesions, abscess/CNS parasitic infections and finally acquired demyelinating disorders , 2.Demyelinating diseases of the nervous system are divided into two major groups based on anatomical localization: demyelinating diseases of CNS and demyelinating diseases of peripheral nervous system (PNS). The major entities in the spectrum of CNS demyelinating diseases are classic multiple sclerosis (MS), MS variants , acute disseminated encephalomyelitis (ADEM), acute hemorrhagic leukoencephalitis (Hurst Disease), optic neuritis, acute transverse myelitis (ATM), and acute cerebellar ataxia (ACA) . The mosSome cases of classic MS, MS variants such as Schilder or Balo disease and some cases of ADEM are the most important acquired demyelinating disorders that can produce tumefactive appearance in brain MRI. Simply, the presence or absence of fever and signs of encephalopathy, previous history of URI (upper respiratory infection) or vaccination as well as brain imaging finding scan help us to differentiate between ADEM and MS variants -8. SchilCorrect diagnosis of the nature of a tumefactive lesion is important, preventing unnecessary brain biopsy, surgery or therapeutic procedures such as radiotherapy or chemotherapy . NowadayHere, we emphasized the MRI finding of Schilder and Balo diseases presenting two real cases.Two patients (two boys with 10 and 8 years old) presented to our hospital as one of the two big referral children hospital in Iran with acute and subacute neurologic complications. Further medical evaluation revealed onset of acute tumefactive demyelinating disease in these two cases. We report clinical and laboratory findings of these two patients in details with a brief review of the role of neuroimaging modalities in how to manage tumefactive lesions in brain MRI.Consent form was written and filled by biologic parents of both two patients to include in our study.Patient 1The first case was a 10-yr-old boy with normal development referred to our hospital with sub-acute onset of right-circumduction gait, some degree of vision loss, expressive aphasia, vomiting and headache started from 1 month ago. There was no history of recent URI or vaccination. In physical examination, he had no fever but right foot drop and right arm weakness in association with bilateral downward plantar reflexes. Fundoscopic examination of the patient showed bilateral papilloedema (grade 2-3). Routine blood chemistries were normal. Brain computed tomography CT scan had been performed elsewhere helped to implement CSF puncture unwarily.CSF analysis showed normal degree of protein, glucose, IgG index, lactate, oligoclonal band (CSF OCB) and negative AQP4 antibody. CSF opening pressure was about 28 cm H2O. Bacterial and viral assessments of CSF and CSF polymerase chain reaction PCR for tuberculosis were all negative. Serum very long-chain fatty acid (VLCFA) levels and adrenal function tests were normal. Initial brain MRI without contrast showed bilateral abnormal high signal lesions in the subcortical and deep posterior white matter in T2-WI sequence that was almost isointense with CSF in the center, surrounded by a peripheral halo . In postPatient 2The second case was an 8-yr-old boy with normal development who presented with acute onset of left-sided hemiparesis of the face and subsequently weakness of left-side limbs following a mild head injury. He had no history of recent viral illnesses or vaccination. Physical examination revealed no loss of consciousness or fever, but he had left-sided limb paresis with hyperreflexia and central left facial nerve palsy. Other neurologic examinations were unremarkable. Brain CT scan with contrast demonstrated low attenuated subcortical areas in the right frontal lobe and right lentiform nucleus with some mass effect .The first brain MRI of the patient showed a couple of hypersignal lesions in T2-WIsequence. The larger one was located in the right lentiform nucleus and insular subcortical white matter with surrounding halo. The smaller one was located in the posterior limb of right internal capsule . Lumbar Virology assessment of CSF for HSV, VZV and HIV were all-negative. The patient was treated with a 4-day course of methylprednisolone pulse with presumptive diagnosis of Balo disease. The clinical symptoms ameliorated, but due to remaining some degree of leftsided limbs weakness, physical therapy was started. About 2 months later, he presented to the Emergency Department with an attack of a febrile seizure. After doing brain CT-scan, the second brain MRI was done that revealed very large mass like concentric or layered high signal lesion in FLAIR sequence in the right frontal lobe white matter and adjacent basal ganglia .A brain biopsy of the right frontal lesion was done and showed a lesion with macrophage infiltration, prominent gemistocytic gliosis and no evidence of malignancy. These findings were compatible with a demyelinating disease and according to neuroradiologic findings, the diagnosis of Balo concentric sclerosis was considered. Carbamazepine was started for the patient and his clinical course followed for 2 yr. The third brain MRI had done 6 months after biopsy, demonstrated volume loss in the right hemisphere with secondary ventricular prominence. Mass-like lesion seen in the second brain MRI in the right frontal lobe could not be seen any longer and abnormal periventricular white matter signal would suggest diffuse gliotic changes or less likely sequel of previous demyelinating lesion . The lasNormally, tumefactive demyelinating lesion is defined as a mass-like lesion with a size greater than 2 cm in brain MRI. Its radiologic features may include mass effect, local edema, ring enhancement and decreased relative cerebral blood volume , 11, 12.Tumefactive demyelinating lesions occur mainly in cerebral hemispheres but may be seen in other parts of CNS, such as cervical spinal segments ,14. BetwThe onset of neurologic symptoms of tumefactive demyelinating lesions may be acute or subacute , 10. TheOne of interesting signs in this patient less reported is speech problem. In addition, the second case  showed hemiparesis and seizure in his clinical course. Increased ICP have been reported in Schilder disease . Schilder and Balo diseases are two rare demyelinating disorders of CNS that may have some similarity in clinical symptoms with each other, but neuroradiologic and pathologic findings help us to distinguish them from each other. The original case of Schilder disease was described by Schilder in 1912 but in 1986 Poser established revised diagnostic criteria for non-invasive diagnosis of true Schilder disease . NeuropaRadiologic features of the first case in brain MRI were compatible with Schilder disease but we did not perform brain biopsy for neuropathologic evaluation. Based on criteria, histopathological evidence is necessary for diagnosis of Schilder disease . In receIn this case, brain biopsy was done initially with probable diagnosis of brain tumor. The pathologic study of brain biopsy of this patient confirmed the diagnosis of Balo disease.It is worth determining the nature of tumefactive demyelinating lesions (TDL) by means of noninvasive diagnostic tools, mainly neuroradiologic tools. Neuroimaging may help to prevent brain biopsy that is an invasive measure and is associated with some morbidity (0.7%-9%) and mortality (0%-1.2%) ,14. In the second case, it was possible to reach the diagnosis of demyelinating lesion without brain biopsy, especially that the clinical symptoms of the patient ameliorated with trial course of steroid and the size of the lesions were decreased in the following brain MRI compare  and 5.Different neuroradiologic measures can help us to characterize the nature of a TDL in brain MRI in a patient with focal neurologic deficit  21-26).-26.21-26These techniques in addition to brain MRI include MRS (Magnetic Resonance Spectroscopy), diffusion- weighted imaging (DWI) and perfusion-weighted imaging (PWI), apparent diffusion coefficient (ADC). Another way that can help to distinguish demyelinating nature of a tumefactive lesion from another differential diagnosis such as an abscess or neoplasm is good clinical response to steroid and reduction of the size of the lesions in serial neuroimaging .In conclusion, demyelinating diseases of childhood have a wide clinical spectrum that can be divided into two groups that involve peripheral or central nervous system (s system ,5. We sus system . In the Ashrafi MR: Designing, Final approval of the work Tavasoli AR: Drafting, Interpretation of the data, final approval of the workAlizadeh H: Neuroimaging interpretationZare Noghabi J: Drafting, Refer one of the two patientsParvaneh N: Draft, Critical reviewAll authors agreed to be accountable for all aspects of the Work in ensuring that questions related to the accuracy or integrity of any part of the work is appropriately Investigated and resolved.


Goblet cell carcinoid is a rare variant of appendiceal carcinoid with mixed endocrine and exocrine features. The most common symptom and signs are abdominal pain, acute appendicitis and palpable mass. Additionally, abdominal pain is common in patient on peritoneal dialysis, which may confound the diagnosis in such patient.We report a 71- years- old woman on peritoneal dialysis that experienced several episodes of abdominal cramping pain and sterile peritonitis. She had one episode of severe pain and underwent an appendectomy for suspicion of appendicitis. Goblet cell carcinoid was diagnosed. She had no further abdominal pain after she received appendectomy.Malignant dialysate was rarely reported in patient with peritoneal dialysis. However, goblet cell carcinoid can initially present with acute appendicitis, chronic intermittent abdominal pain and mimicking peritonitis. In systemically reviewing the literature, this is the first case report of sterile peritonitis with peritoneal dialysis caused by goblet cell carcinoid. Goblet cell carcinoid is a rare variant of appendiceal carcinoid with mixed endocrine and exocrine features. The most common clinical presentation is acute appendicitis , 2. TypiA 71- years- old woman with underlying hypertension and end- stage renal disease on peritoneal dialysis, experienced several episodes of abdominal cramping pain with mild diarrhea 3 months after starting peritoneal dialysis. She had chills but no fever. A physical examination showed lower abdominal tenderness, but no rebounding pain. Initial complete blood count showed no leukocytosis (8400/μL). However, the initial dialysate studies showed increasing leukocytes with a predominance of neutrophils in first few sample, shifting to lymphocyte predominance in later sample , an unique and enigmatic tumor involving the appendix almost exclusively, is a rare variant of appendiceal carcinoid . CarcinoThe most common clinical presentation is acute appendicitis . AbdominThe most common route of metastases is trans-coelomic/peritoneal invasion, and the most common sites involved are ovaries, and the peritoneal surfaces of the pelvis and abdominal cavity .The natural history of patients with GCC is intermediate in aggressiveness between adenocarcinomas and classical carcinoids . Based oOur patient, who experienced abdominal pain intermittently for months had been diagnosed with peritonitis related to peritoneal dialysis. Given a clinical history of recurrent sterile peritonitis with a predominance of lymphocytes in the dialysate, an atypical infection (including tuberculosis) and intra-abdominal malignancy must be considered in the differential diagnosis. However, no dialysate or ascites was sent to cytology analysis in our patient. We also considered intermittent bowel obstruction as the cause of abdominal pain, which was the common presentation of GCC , but theOf all the carcinoid tumors, GCC, in particular, can initially present with acute appendicitis, chronic intermittent abdominal pain, and malignant ascites mimicking peritonitis related to peritoneal dialysis in such patient. Through systematic review of PubMed and Cochrane Library databases, there were no similar cases reported. We report this unique case of GCC presenting with sterile peritonitis in a patient on peritoneal dialysis to prompt clinicians to consider GCC in the differential diagnosis of similar symptoms in such patients.


Esophagogastroduodenoscopy performed six years after the diagnosis revealed multiple erosions in the gastric body and angle. Magnifying endoscopic observation with narrow-band imaging showed that the gastric pits were only partially preserved and were destroyed in most of the stomach. Branched abnormal vessels were also seen. Pathological features were consistent with follicular lymphoma in both cases. The structural differences reported between the two cases appear to reflect distinct pathologies. Disappearance of gastric pits in the latter case seems to result from loss of epithelial cells, probably due to chronic inflammation. In both cases, branched abnormal vasculature was observed. These two cases suggest that magnified observations of abnormal branched microvasculature may facilitate endoscopic detection and recognition of the extent of gastric involvement in patients with follicular lymphoma.A 70-year-old woman presented with follicular lymphoma involving the stomach, duodenum, jejunum, bone, and lymph nodes. Esophagogastroduodenoscopy revealed multiple depressed lesions in the stomach. Examination with magnifying endoscopy showed branched abnormal vessels along with gastric pits, which were irregularly shaped but were preserved. The second case was a 45-year-old man diagnosed with stage II Follicular lymphoma is the second most frequent subtype of lymphoid malignancies observed in western countries. In patients with follicular lymphoma, the gastrointestinal tract can be primarily or secondarily involved . Most gaRecently we experienced two cases of systemic follicular lymphoma involving the stomach. This paper focuses on the pathologic and endoscopic features of gastric lesions of follicular lymphoma. We also speculate on the pathophysiological processes behind the microstructural findings in both presented cases. Helicobacter pylori infection. Biopsy samples from the peripheral intact mucosa had no neoplastic cells, whereas samples from the gastric lesions showed dense, diffuse infiltration of small- to medium-sized lymphoma cells  showed slightly depressed lesions with thickened peripheral folds in the upper  and lowema cells . LymphoeVideo capsule enteroscopy showed whitish granules in the jejunum . Compute1 follicular lymphoma with duodenal, ileal, colorectal involvement and lymphadenopathy of the mesenteric lymph nodes. Lymphoma progression with axillary and inguinal lymph node swelling was noted one year later, but the patient was kept under active surveillance . Two years after the initial diagnosis, gastric involvement was diagnosed. Esophagogastroduodenoscopy performed six years after the diagnosis revealed whitish depositions in the duodenum ( Helicobacter pylori titer was negative in this patient. Biopsy samples from the gastric lesions showed a diffuse infiltration of small- to medium-sized lymphoma cells, in addition to the existence of granulation tissue and lymphoid follicle (Figures A 45-year-old man was diagnosed with stage IIduodenum . Multiplduodenum . No erosduodenum . Howeverduodenum , whereasduodenum . Branche Figures . Immunoh Figures , CD10 (F Figures , and BCL Figures . Stainin Figures  in the n Figures . In cont Figures . Esophag Figures . BecauseLymphoma derived gastric lesions present with diverse endoscopic features, varying from mass-forming tumors to diffuse infiltrating lesions or superficial mucosal changes . MoreoveAlthough gastric involvement is infrequent in follicular lymphoma patients, it is important to evaluate the gastric mucosa during both the initial evaluation and the follow-up period as part of a thorough diagnostic examination. During the initial diagnostic workup of follicular lymphoma patients, determination of the extent of disease present is recommended for stage I and II patients who are being considered for radiotherapy as a curative treatment. Staging in these patients is important since disease relapse tends to occur outside the involved field of radiation . Even th Helicobacter pylori eradication, abnormal vessels were no longer detected and gastric pits reemerged, though those pits were irregularly patterned and had a different form and a different density from those in the adjacent intact mucosa. Consequently, unusual-shaped vasculature, a nonstructural pattern, and destruction of gastric pits appear to be specific for untreated gastric MALT lymphomas.To the best of our knowledge, magnifying endoscopic features observed in gastric involvement of follicular lymphoma have not been reported to date. As described above, involvement of the stomach is more frequent in MALT lymphoma, which has been a well-recognized entity among lymphomas involving the stomach since first described by Isaacson and Wright in 1983 . Typical H. pylori-associated chronic gastritis and diffuse type of gastric cancer [In both of the present cases, magnifying endoscopic observations with narrow-band imaging showed abnormal branched vessels. As described above, branched microvasculature observed in our cases was similar to the reported features in cases of MALT lymphoma . Moreovec cancer , 27. Thec cancer . Second,In case 1, the gastric pits were found in an irregular pattern but were nonetheless intact. In contrast, case 2 exhibited diffuse destruction of gastric pits with only limited sections showing intact pit structure. We speculate that the structural differences between the two cases reflect distinct pathologies. The destruction of gastric pits observed by endoscopy in case 2 seems to result from the loss of epithelial cells . On the In conclusion, we treated two patients with follicular lymphoma involving the stomach. Abnormal branched microvessels were observed in both cases, whereas gastric pits were preserved in one patient and destroyed in the other. Although appropriate pathological assessment with immunohistological analysis of biopsy samples is essential for definitive diagnosis, magnified endoscopy of microvascular structures and gastric pits may be useful in alerting physicians to the potential for lymphoma lesions in the stomach.


Angioedema (AE) classically manifests as an acute transient swelling of extra-visceral spaces, subcutaneous and submucosal tissues. Sometimes it may be a life-threatening condition. The causes are numerous, and the common denominator is an increased vascular permeability allowing diffusion or extravasation of fluid from the vascular bed to the interstitial space. The severity of AE is related to the cause, body location, and extension. We hereby report two very unusual cases characterized by a massive attack of AE from the left cervical area to the pelvis through the length of the mediastinum and axial posterior retroperitoneum. The diagnosis was established by CT. The first case was found related to drug intake, and the second appeared idiopathic. Angioedema (AE) – first named angioneurotic oedema – is an acute transient phenomenon due to increased vascular permeability allowing diffusion or extravasation of fluid from the vascular bed to the interstitial space . It resuThe result is classically an acute swelling of extra-visceral spaces, subcutaneous and submucosal tissues lasting two to seven days. In some cases, it may be a life-threatening condition. Severity of AE is related to the cause, body location, and extension. Superficial regions in the face, neck, and upper airways are mostly involved. More rarely, AE may manifest by visceral AE involving the abdominal organs and especially the gastrointestinal tract. Genitourinary involvement may also be rarely encountered 34.3We hereby report two very unusual cases drastically differing from all classical presentations and in which a massive attack of AE was found extending from the neck to the pelvis through the length of the mediastinum and the axial posterior abdominal retroperitoneum. The full diagnosis was made by CT in the two cases. The first was considered related to drug intake and the second seemed idiopathic.A 45-year-old woman presented to the emergency department with marked clinical swelling of the left supraclavicular fossa. The phenomenon had suddenly developed within a few hours and without any particular effort nor cough or pain. The patient also reported some chest tightness. Physical examination revealed slight left paraombilical tenderness. Basic laboratory tests were irrelevant.On contrast-enhanced thoraco-abdominal CT Figure , massiveComplementary laboratory tests excluded acquired or congenital deficiency of the C1 esterase inhibitor (C1-INH). A possible relation was found with the intake of Biocondil by the patient. Biocondil contains chondroitin and glucosamine, this last component being synthesized from chitin extracted from the crustacean carapace , a well-known allergenic potential.A 60-year-old woman was referred to the emergency department with an acute swelling of the left supraclavicular and left laterocervical areas. These complaints were associated with intense abdominal pain. Basic laboratory tests were irrelevant. Anamnesis revealed a previous and spontaneously resolving episode of ascite and left cervical swelling 15 years earlier.Cervical ultrasound showed a major dedifferentiation and oedema of the left laterocervical of supraclavicular areas. A heterogeneous tubular structure penetrating the distal internal jugular vein was first considered as a venous thrombosis Figure . ComplemThe tubular structure identified during ultrasound at the junction of the internal jugular and subclavian vein Figure  was cleaIt was first concluded that the common causal denominator of the two episodes of AE was the intake of diclofenac ®, but another episode of AE occurred spontaneously a few weeks later, nullifying this hypothesis. The AE was thus considered idiopathic, and the patient is now preventively empirically treated with Tranexamic acid (Exacyl ®).The physiopathological causes of AE are numerous but may pragmatically be classified following their main mechanism resulting from either bradykinin or histamine mediated responses 2.Bradykinin-mediated AE disorders essentially comprise the hereditary AE (HAE) due to a decrease level or a dysfunctional C1 esterase inhibitor (C1-INH), secondary acquired C1 inhibitor deficiency (associated with lymphoproliferative or autoimmune diseases), and specific types induced by various drugs. Angiotensin-converting enzyme (ACE) inhibitors are mostly involved, but also many other drugs. Bradykinin-mediated AE is not associated with urticaria and not mediated by IgE antibodies.AE mediated by histamine mostly concerns allergic or idiopathic AE – which represents the majority of cases and present with or without urticaria or anaphylaxis. When a trigger is identified, it is most often a drug, an alimentary allergen, latex, or an insect sting . A physiIn our first patient, the cause appeared related to drug intake, but no cause was established for the second, which was considered fully idiopathic.AE have a longer duration 2–5 days) when mediated by bradykinin than histamine 24–48 hours), and classically the first do not respond to antihistamines and corticosteroids . Clinica–5 days w3–48 hoursThe role of medical imaging is very limited in these types of self-limiting peripheral cases of AE classically involving the face, the neck, and also but more rarely external genital organs or the extremities . MedicalMore rarely and sporadically, AE may also involve deep organ systems such as the gastrointestinal tract and the genitourinary tract, even in the absence of peripheral cutaneous involvement and thus without external visible manifestation. The acute or recurrent symptoms are non-specific, comprising severe abdominal pain with distension and tenderness, nausea and vomiting, diarrhoea or sub-occlusion. Acute pancreatitis may be associated due to duct obstruction or oedema of the ampulla of Vater.Medical imaging is more valuable in these cases 3456. Con3452In typical cases of visceral AE, the affected bowel loops show a typical target or “halo” sign due to a low-density mucosa oedema contrasting with mucosal or subserosal enhancement 6. Free aGenitourinary involvement may also be rarely encountered. Renal involvement may simulate renal colic and urinary bladder AE presents with lower abdominal pain and retention. The cardinal signs are oedematous wall thickening with adjacent fluid 4.Our two reported cases drastically differ from all these classical presentations, and to our best knowledge, such massive attacks of “total body AE” have not been clearly reported before, even in a recent review .We have only identified a single case report of isolated and relatively dominantly elective retroperitoneal ACE inhibitor–induced AE . The CT Classically retroperitoneal oedema has only sometimes been reported as an associated finding of gastrointestinal visceral AE, but not as an isolated event, as in our patients . An inteThis type of swelling was reported by Dendorfer  as a recA heterogeneous pseudocystic dilatation of the cervical distal portion of the thoracic duct was observed in our second patient. Nevertheless, this entity was transient and completely disappeared – CT had normalized four days later – thus rejecting the hypothesis of malformation or obstruction of the duct. Moreover, the fact that the full length of the thoracic duct as well as the cisterna chyli were simultaneously dilated could probably be the result of overflow of the lymphatic system caused by the massive mediastinal and retroperitoneal fluid extravasion.The reports of our patients also drastically differ from the clinical and imaging features resulting from situations of disruption or blockage of the thoracic duct or of its main lymphatic tributaries. The causes of these situations may be tumoral, mechanical but also surgical or traumatic 101112. T1011To conclude, we reported two rare occurrences of extensive AE. It may be questionable whether the two reported cases are really quite rare and atypical – because of their unusual extensive thoraco-abdominal manifestations – or if they simply correspond to situations that are more frequent but overlooked because of insufficient imaging. Indeed, the dominant clinical symptom of our two patients was a cervico-supraclavicular swelling, thus a mainly peripheral symptom likely to be under-investigated.


To the Editor,Intravascular large B-cell lymphoma (IVLBCL) is a rare type of extranodal B-cell lymphoma characterized by the growth of lymphoma cells within the lumina of small vessels. Two major patterns of clinical presentation have been recognized: the first is in European countries, with brain and skin involvement, and the second in Asian countries, where patients typically present with multiorgan failure, hepatosplenomegaly, pancytopenia, and hemophagocytic syndrome ,2,3,4,5.max: 7.0) and multiple lymph node lesions including the submandibular, preauricular, cervical, and jugular lymph nodes (SUVmax: 8.3). He was treated with six cycles of an R-CHOP regimen. He did not show any evidence of recurrence  at 36 months of follow-up.A 60-year-old male patient was admitted to the hospital with fever, abdominal pain, and weight loss. Physical examination showed an epigastric mass of approximately 4 cm in diameter and the absence of hepatosplenomegaly and lymphadenopathy. Laboratory tests revealed anemia (hemoglobin: 10 g/dL), with normal leukocytes and platelets. Peripheral smear showed normocytic-normochromic anemia without any abnormal cells. Increases in liver function tests were positive laboratory findings . Contrast-enhanced abdominal computerized tomography (CT) for further evaluation revealed a greater curvature-based mass of 8x5x5.5 cm in size, at the level of the distal gastric corpus, significantly narrowing the gastric lumen . CT alsoIVLBCL usually occurs in adults in the sixth and seventh decades. The tumor is often clinically unsuspected and can be easily overlooked on biopsy. The diagnosis is most commonly made at autopsy. The lymphoma cells are generally large with round nuclei and prominent nucleoli. The malignant cells uniformly express pan-B-cell antigens CD20, CD79a) and variably express other antigens such as CD5 (38%) and CD10 (13%) [0, CD79a