Emphysematous cholecystitis is a severe variant of acute cholecystitis caused by anaerobic bacteria. Although intraperitoneal air as a complication has been described in association with emphysematous cholecystitis, pneumoretroperitoneum arising from emphysematous cholecystitis is extremely rare. Herein, we describe a rare case of pneumoretroperitoneum arising from emphysematous cholecystitis that was successfully treated with emergency surgery.Klebsiella pneumoniae, which was considered to be the cause of emphysematous cholecystitis, pneumobilia, and pneumoretroperitoneum.An 84-year-old male was transported to the Emergency Department of our hospital for acute abdomen. Computed tomography revealed acute cholecystitis accompanied by emphysematous change. Computed tomography also revealed massive pneumoretroperitoneum complicated with pneumobilia and gas in the hepatoduodenal ligament. Clinical findings fulfilled the diagnostic criteria for systemic inflammatory response syndrome and sepsis. Emergency surgery was carried out with a diagnosis of both emphysematous cholecystitis and gastrointestinal perforation. Intraoperative findings revealed acute gangrenous cholecystitis and pneumoretroperitoneum presenting with an odor-free foamy abscess along the loose connective tissue behind the ascending colon and mesocolon. No evidence of gastrointestinal perforation was found during surgery. Therefore, cholecystectomy and lavage drainage were performed. Bacterial culture examination isolated a single species of anaerobe, Emphysematous cholecystitis should be considered as a possible cause of pneumoretroperitoneum. The present case is the first report of massive pneumoretroperitoneum extending to the dorsal side of the ascending mesocolon as a complication of emphysematous cholecystitis. Escherichia coli, Aerobacter aerogens, Klebsiella spp, Clostridium spp, and Salmonella spp, leading to severe complications: gangrene, perforation of the gallbladder, and pericholecystic abscess [Emphysematous cholecystitis (EC) is a rare, severe, potentially lethal variant of acute cholecystitis characterized clinically by gas accumulation in the wall and lumen of the gallbladder or pericholecystic tissue , 2. It i abscess , 4. The  abscess .Extra-intestinal gas, which strongly suggests perforation of the gastrointestinal tract, is a critical sign of the need for emergency surgery. Previous reports have indicated that EC leads to the development of intraperitoneal air over the progression of anaerobic infection , 5. Howe2 of 25.5 mmHg . These clinical findings fulfilled the diagnostic criteria for systemic inflammatory response syndrome and sepsis. An abdominal X-ray showed irregular gas accumulation in the right lateral abdomen  with 91.0 % neutrophils, hemoglobin of 15.4 g/dL , glycated hemoglobin A1c (HbA1c) of 6.9 %  , fasting blood sugar of 195 mg/dL , aspartate aminotransferase of 23 IU/L , alanine aminotransferase of 12 IU/L , alkaline phosphatase of 249 IU/L , total bilirubin of 1.2 mg/dL , γ-guanosine triphosphate of 17 IU/L , serum creatinine of 1.23 mg/dL , and C-reactive protein of 5.93 mg/dL . Urinary analysis showed 1+ glucose and 3+ bacteria. Arterial blood gas analysis showed respiratory alkalosis and decreased pCOMost ECs occur in patients aged 50 ~ 80 years . Unlike In the diagnosis of cholecystitis, ultrasound is indeed available and widely conducted for detection of earlier stage cholecystitis. With regard to EC, the previous reports indicated that the air in the wall and lumen of the gallbladder interfered in visualization of ultrasound image with a reverberation artifact . BesidesExtra-luminal gas complicated with EC has been conventionally considered to derive from gallbladder perforation . WhenevePneumoperitoneum and pneumoretroperitoneum as complications, which represent extended infections, seem to be obviously more severe condition compared with EC localized in the gallbladder. Gill et al. divided EC radiographically into three stages according to the distribution of air within the gallbladder and/or the biliary system as follows: Stage1 –air in the gallbladder lumen, Stage2 –air in the gallbladder wall, and Stage3 –air in the pericholecystic tissue . AccordiProgression of EC can cause the spread of gas to the hepatoduodenal ligament  or escapEC should be considered as a cause of pneumoretroperitoneum. Whenever we encounter pneumoretroperitoneum concurrently with EC, it should be assumed that retroperitoneal gas might have derived from gas-producing anaerobic infection of the gallbladder extending to retroperitoneum. The present case is the first report of massive pneumoretroperitoneum extending to the dorsal side of the ascending mesocolon as a complication of EC.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


We report multiple synchronous clear-cell chondrosarcomas in a 43-year-old patient. The patient had a lesion in the right proximal humerus and in the left femoral condyle. Bone scintigraphy revealed increased uptake in both foci. Pathological analysis confirmed the diagnosis in both locations. In the proximal humerus, wide resection of the tumour was performed with allograft reconstruction of the joint with osteosynthesis. The femoral condyle was treated with curettage, phenolization, and cementation. Over a follow-up of 10 years no recurrence or metastasis was observed. Clear-cell chondrosarcoma (CCC) was first described by Unni et al.  as a varThe tumor is most frequently found in the epiphysis of the long bones . Most authors agree that the management of choice is wide resection. The estimated survival rate at 5 years is 92% . MetastaHere we report a case of synchronous CCC with a favorable outcome at 10 years.A 43-year-old male patient was seen with a one-year history of pain in the right shoulder and limited range of motion of the joint. Radiographs revealed a lytic lesion in the proximal epiphyseal humerus, with calcifications, and cortical thinning and expansion of the bone Figure a. ComputCT scan of the thorax was normal and alkaline phosphatase within normal limits.Needle biopsies were performed and the diagnosis of CCC was confirmed in both sites Figures d and 3d.CCC is a rare low grade variant accounting for 2% of all chondrosarcomas ,5,6. TheThe condylar lesion had clinical and imaging features that were similar to epiphyseal chondroblastoma; but the patient was too old, and there was no inflammatory reaction on MR,as usually seen in chondroblastoma. Some authors suggest that CCC may be a malignant chondroblastoma ,12. AchiWe found no multiple synchronous or not CCC reported in the literature . RecurreUnni et al.  reportedUnfortunately, in our case previous radiographs of the knee were not available. As in this location the lesion was small, asymptomatic, and was an incidental finding on the bone scintigraphy while physical examination was normal and the imaging findings indicated a non aggressive lesion, it was considered to be an inactive lesion. The fact that the patient had no other metastasis in the follow-up is also in favor of two primary lesions, even if formal proof cannot be established. Due to the slow growth of the tumor, long-term follow-up is essential in CCC, as local recurrence more than 20 years post-operatively has been described.Written informed consent was obtained from the patient . Approved by the ethical committee of the Rizzoli Institute.The authors declare that they have no competing interests.MM operated and followed the patient, proposed the subject. SF wrote the article. AR provided and checked the histology. JM checked the article. DV provided the images, and supervised the article. All authors read and approved the final manuscript.


A lipoma is a common, benign soft-tissue tumor that rarely arises in the upper limb. When one does occur in the hand, the location of the lipoma can cause nerve compression, which can mimic carpal tunnel symptoms. Magnetic resonance imaging is the visualization modality of choice for diagnosis and surgical planning of lipomas. Surgical resection is recommended to relieve the neurological manifestations of this disease. The surgeon should always suspect liposarcoma first before voluminous, atypical, or recurrent tumors are considered. Lipomas are the most common benign tumors found in limbs . AlthougA 69-year-old right-handed woman presented with a gradual tumefaction on the palm of her right hand that developed without any traumatic or microtraumatic antecedent.Interrogation of the patient found a progressive evolution over several months with gradual acroparesthesia in the median nerve territory associated with palmar pain. Physical examination discovered a decrease in epicritic sensitivity with the Weber test, without evidence for motricity impairment, using preserved prehensile strength and digital winding as readouts.Palpation revealed limited renitent tumefaction on the palm of the hand, without cutaneous suffering. There were no signs of septic diseases  or tumoral dissemination .Functional exploration with electromyography identified a sensitive injury downstream of the carpal tunnel. Standard X-rays did not found abnormalities. Magnetic resonance imaging (MRI) found a large tumor, which was homogeneous, multilobulated, and with a sharp border. The tumor had an intense hypersignal in T1 and a poor fat suppression signal Figures . The lesResection was performed under locoregional anesthesia with an axillary block. The patient was arranged in a dorsal decubitus position; a pneumatical tourniquet was placed around the arm and inflation was maintained during the surgical procedure.A palmar zigzag approach through palmar-crease was chosen face up of the tumor to promote release of the neurovascular pedicle joining the tumor to the hand . Lipoma Postoperatively, the patient went home with scar care every other day and a prophylactic treatment for algoneurodystrophy with vitamin C for one month. No immobilization was used.Complete healing occurred after 14 days. The last clinical review at the one-month postoperative clinical examination showed entire disappearance of the acroparesthesia and total functional recovery including full mobility of the fingers, absence of pain, and restoration of the grip strength. No specific rehabilitation was necessary.Histopathological analysis concluded that the lesion was a lipocytic lipoma with no evidence of malignancy.Lipomas represent 16% of all mesenchymal tumors  and are Lipomas of the hand principally occur in individuals between 50 and 60 years of age , 4. PatiIts clinical expression is identified by the topography of the compression: carpal tunnel syndrome in the case of median nerve compression , 5, hypoGenerally, standard X-ray examinations do not aid in the diagnosis because they only show a homogeneous opacity in the soft tissue. However, sometimes they reveal calcifications in the tumoral body or a cortical bone condensation, in the case of parosteal lipoma .Ultrasound is an effective tool to explore any mass existence in the hand. Physicians easily request ultrasound in case of atypical symptoms of carpal tunnel syndrome. Ultrasound provides a view of the anatomy of nerve as well as of surrounding structures and allows real-time and painless assessment of median nerve entrapment. Typical findings associated with nerve compression include enlargement of the nerve proximal to the site of compression, decreased echogenicity, and increased vascularity . MoreoveMRI is the visualization modality of choice for exploration of tumors of the hand. Primarily, it allows a diagnostic orientation of the tumor, and, secondly, it shows anatomical relationships between the tumor and the neurovascular structures. Classically, lipomas appear as a homogeneous mass, with a sharp border, spontaneous T1 and T2 hypersignals, reduced signal intensity after erasure of the fat signal, and no raising in the signal using the gadolinium contrast agent , 13. FinWe advocate for a thorough radiographic examination in all cases of atypical clinical presentation of carpal tunnel syndrome including unilateral symptoms, sudden onset, unusually young patient, and clinical mass syndrome.Differential diagnosis for this type of tumours includes principally fibrolipoma of the median nerve  and low Liposarcoma remains the most prevalent diagnosis , particuRarely, other differential diagnoses occur and could be another tumor of the soft tissue, such as a ganglion cyst or giant-cell tumor . LeiomyoThe size of 5 cm is the classical cutoff for definition of giant lipoma , 21, 22.Therapeutically, surgical resection of lipomas is the single treatment that allows liberation of the compressed nerve endings and effectively removes the tumor. Surgeons should perform a monobloc resection and a careful, safe dissection of the neurovascular branches to maximally reduce the risk of iatrogenic lesions. Local recurrences are rare with histological confirmation of the benign nature of the lipoma. However, some authors assert that the risk of recurrence correlates with the degree of heterogeneity found in the first MRI examination .The typical or atypical manifestation of nerve compressions in the hand should be investigated for a tumor syndrome in a clinical examination. Practitioners should then try to link the tumor localization with the nerve manifestations. Functional exploration with electromyography is indispensable for authentication of the level of compression. Next, MRI is the best morphological procedure, in terms of predictive values, to explore tumors of the hand and wrist. The size of 5 cm should be the limit for definition of a giant lipoma for hands. Moreover, this size is important to define the level beyond which practitioners must first perform a biopsy to exclude the risk of liposarcoma before complete resection. Surgery is the modality of choice to treat such lesions and results in a good functional recovery. The primary risk remains the risk of iatrogenic lesions during surgical resection, caused by the intimate proximity between the lipoma and neurovascular endings.


Spontaneous intraspinal intramedullary hemorrhage is a rare entity with the acute onset of neurologic symptoms. The etiology of idiopathic spontaneous hematomyelia (ISH) is unknown, and there are few published case reports. Hematomyelia is mostly associated with trauma, but the other nontraumatic etiologies are vascular malformations, tumors, bleeding disorders, syphilis, syrinx, and myelitis. MRI is a good choice for early diagnosis. Hematomyelia usually causes acute spinal cord syndrome due to the compression and destruction of the spinal cord. A high-dose steroid treatment and surgical decompression and evacuation of hematoma are the urgent solution methods. We present idiopathic spontaneous hematomyelia of a previously healthy 80-year-old male with a sudden onset of back pain and paraplegia. The ISH is a rare entity. Trauma is considered the most common cause of hematomyelia. The other etiologies are vascular malformations, cavernomas, aneurysms, primary tumors or metastasis, syrinx, meningomyelitis, myelitis and arachnoiditis, hemophilia, Von Willebrand's disease, factor 11 deficiency, anticoagulant therapy, and other rare causes including syphilis and aortic aneurysms . SurgicaAn 80-year-old man developed sudden severe back pain with no history of trauma, and then he tried to walk. However, due to weakness and numbness in his legs, he could not walk and collapsed to the ground. He presented to an outside hospital with paraplegia, and then he was transferred to a tertiary center for further management. On admission, neurological examination of his motor power was reduced (0/5) in both lower extremities (paraplegia), tendon reflexes in both lower limbs had increased, Babinski responses were positive, there was anesthesia/hypoesthesia for light touch, and there was hypoesthesia below the thoracal (T5-T6) dermatome. His knee- and ankle-jerk reflexes were absent bilaterally. There were urine and stool incontinence. The complete blood count , biochemistry profile , prothrombin time, and INR were normal, and he did not have additional comorbidities . In addition, he did not use an anticoagulant therapy. At the time of admission to emergency, only subcutaneous low molecular weight heparin (0.3 mL) was conducted. A high-dose steroid treatment was given on the day of his admission (bolus 30 mg/kg administered over 15 minutes with maintenance infusion of 5.4 mg/kg per hour infused for 23 hours).The Computed Tomography (CT) scan was not diagnostically helpful. Magnetic resonance imaging (MRI) revealed an intramedullary mass with signal intensity surrounded by a zone of high and low signals on both T2 weighted images Figures . UnfortuThe patient underwent the operation on the day of admission. After thoracic vertebra 5 and 6 laminectomy, microscopic dural opening was revealed, a small paramedian myelotomy was performed at the level, and blunt dissection and aspiration of the hematoma were done. A hematoma was completely removed . There wHematoma in the spinal cord is called hematomyelia. Spontaneous hematomyelia is a rare entity. The etiologies are trauma, vascular malformations , primary tumors or metastasis, and parenchymal abnormalities, such as syrinx, meningomyelitis, myelitis and arachnoiditis, or spinal radiation changes, and bleeding disorders . Other rare causes include syphilis, aortic aneurysm, hemorrhagic myelitis or hyperdynamic states of spinal artery circulation, and cardiovascular disorders . Trauma is considered the most common cause of hematomyelia , 6–9.The idiopathic hematomyelia's real etiology is unknown –5, 9. InMRI examination is a better diagnostic tool than conventional myelography or CT. The final diagnosis of ISH is often confirmed by microscopy , 6.Early diagnosis and surgery are essential for cases of ISH in the literature review. Previously reported cases of idiopathic spontaneous intraspinal intramedullary hematoma presented with acute, subacute, or chronic myelopathy. Acute hematomyelia is often accompanied by sudden severe back or neck pain and then presents with myelopathy syndromes due to the mass effect's acute obstruction of CSF and blood in the spine. Hematomyelia usually presents with an acute onset and rapid deterioration in neurologic status . PrognosThere are currently no guidelines for the management of acute hematomyelia, and treatment is typically directed toward the underlying cause .The conservative management is unknown in idiopathic hematomyelia; therefore, surgical removal of hematoma is the treatment of choice for acutely deteriorated idiopathic hematomyelia .Early recognition, accurate diagnosis, and appropriate treatment can decrease morbidity and improve the prognosis and outcome for patients with IMH , 5.Even though high-dose steroid treatment, early decompression, and hematoma microdissection were performed, our patient's neurological examination was unchanged. We should conduct the operation as early as possible, but, first, physical and neurological examination must be done at an emergency department. We must eliminate the blood tests for cardiac or pulmonary emergencies, and then CT and MRI should be done, and when we discover the pathology, we should request the patient's or his family's permission; all these levels took time. We were in the theater room in the 16th hour of the patient's first admission.Given the limited number of similar reports, whether the stabbing pain preceded the intramedullary hemorrhage remains to be determined –10.The present case report clearly describes the spontaneous occurrence of hematomyelia without demonstrable predisposing or precipitating cause, which is not clearly established in the literature. The patient suffered acute and severe neurological deterioration and has limited postoperative functional recovery as a result of the hemorrhage. Given the devastating clinical course of our patient, it is important to recognize the possibility of spontaneous spinal intramedullary hemorrhage.Nontraumatic idiopathic spontaneous intraspinal intramedullary hemorrhage is a very rare entity. Intramedullary hemorrhages are catastrophic. Early recognition, diagnosis, medical treatment, and urgent surgery can decrease morbidity. Using high-dose steroid treatment and microscopic hematoma evacuation may decrease morbidity.


Adrenocortical carcinoma is a rare and aggressive malignancy. Patients usually present early with manifestation of abnormal hormone secretion. However, adrenocortical carcinoma can also be nonfunctioning, and such patients present late with a mass effect or distant metastases.We herein report a case of a 30-year-old Sri-Lankan woman who presented with a 3-month history of left flank pain associated with nausea, vomiting, and weight loss. Imaging revealed a large left upper quadrant mass with a 1.8-cm left lung nodule. The differential diagnoses included a left adrenal mass, left upper pole renal mass, and retroperitoneal sarcoma. A functional adrenal work-up revealed no abnormal findings. Surgical excision of the mass was uneventful with no postoperative complications. Pathological analysis revealed a nonfunctioning adrenocortical carcinoma measuring 16 × 14 × 10 cm. To our knowledge, a mass of this size is among the largest nonfunctioning adrenocortical carcinomas reported in the published literature. The investigations and approach to treatment were consistent with those in the published literature.Large nonfunctioning adrenocortical carcinomas pose a diagnostic and therapeutic challenge, and most are diagnosed at a late stage. Appropriate imaging and functional work-up of the mass are vital before treatment. Surgical excision is safe, even for large adrenocortical carcinomas; excision in patients with advanced disease has been shown to have the best outcomes. Adrenocortical carcinoma (ACC) is a rare and aggressive malignancy with an estimated annual incidence of one to two cases per million inhabitants in the United States , 2. The Adrenal masses can be broadly divided into hyperfunctioning (hormone-secreting) and nonfunctioning (non-hormone-secreting). Functioning ACC presents earlier with hormonal manifestations such as virilization, feminization, or Cushing’s syndrome. However, nonfunctioning tumors pose a diagnostic challenge because they are diagnosed incidentally due to a mass effect or metastatic disease. Moreover, successful radical management is seldom achievable because most diagnoses are made when the tumor has either invaded local structures or metastasized, explaining the poor prognosis associated with these masses , 3.Even after a grossly complete resection, most patients develop early tumor recurrence or distant metastasis . This isThe most frequent metastatic sites are the lung, lymph nodes, liver, bone, and inferior vena cava at 71%, 68%, 42%, 26%, and 20%, respectively . We hereA 30-year-old Sri-Lankan woman presented with left flank pain. The pain was intermittent, sharp, and of moderate severity and radiated to the groin. The patient reported no history of lower urinary tract symptoms or gross hematuria. The pain was associated with weight loss of 8 kg over a period of 3 months. Her medical and surgical history was not significant, and the patient was not on any regular medications.The patient was afebrile on presentation with no microscopic hematuria. On physical examination, the abdomen was soft with minimal tenderness over the left flank, and a palpable left upper quadrant mass was present. The rest of the clinical examination revealed normal findings.A computed tomography (CT) scan showed a large left upper quadrant mass measuring 17 × 13 × 12 cm in its greatest dimension. The mass crossed the midline and compressed the stomach, spleen, pancreas, and small bowel  showed evidence of bone metastasis. A decision was made to proceed with en bloc excision of the mass. Cardiothoracic surgeons were consulted regarding the suspicious lung nodules, and they planned to biopsy the lung lesion after excision of the primary tumor.®) was used to ensure adequate exposure. The mass was exposed after medial reflection of the descending colon and was found to be displacing the stomach, pancreas, and spleen, but it had a grossly negative margin with no evidence of adjacent organ invasion. The mass was covering the left renal hilum and was densely adhered to the aorta. After adequate exposure of the left kidney, the mass was noted to be densely adhered to the upper pole with no surgically defined plane. It seemed to be arising from the upper pole of the left kidney. In the presence of a vascular surgeon, the mass was dissected and the left renal hilum was carefully isolated. We decided intraoperatively to proceed with left radical nephrectomy. This decision was made to avoid positive surgical margins if partial nephrectomy was performed given the wide base of tumor adherence to the kidney. Upon vascular control of the left renal artery and vein, the mass significantly shrunk in size and became less congested. It was then easily dissected off the aorta. No enlarged lymph nodes were identified. After ensuring adequate hemostasis, Surgicel® Fibrillar™ was applied, and an intra-abdominal drain was inserted.A Chevron incision was made, and a Gomez poly tract retractor  is superior to CT alone . HoweverWith the presence of a questionable metastatic pulmonary nodule and large tumor of >4 cm, the clinical suspicion of malignancy was high in this case and favored radical adrenalectomy –14. To tDespite the poor prognosis in this patient group, chemotherapy has a limited role in the treatment of ACC, and surgical resection has been shown to have the best outcomes , 8. HoweWritten informed consent was obtained from the patient for publication of this Case Report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


We reported a rare case of hypothalamic amenorrhea and transverse vaginal septum. A 28-year-old woman presented with primary amenorrhea and no complaint of abdominal pain. Laparoscopy revealed a small rudimentary uterus with streak ovaries and a vaginal pouch. The patient with diagnosis of Mayer-Rokitansky-Kuster-Hauser (MRKH) syndrome was subjected to a vaginoplasty in another fertility center. In our institute, after two courses of estrogen and progesterone, sonography revealed hematocolpos, while, under anesthesia, transverse vaginal septum was resected. Hysteroscopy revealed normal uterine cavity. She became pregnant 5 months postoperatively with controlled ovarian stimulation (COS) in conjunction with intrauterine insemination, and she has two healthy babies now. This case highlights the importance of careful evaluation of all primary amenorrheas. Clinicians should be aware of presence of more than one etiology which causes atypical presentations and accomplishes a systematic strategy for the evaluation of amenorrhea potential to avoid long-term side effects of a misdiagnosis. The recommended evaluation for amenorrhea aimed to divide the reproductive system into its components—the genital outflow tract and uterus, the ovary, the pituitary, and the hypothalamus—and to assess the functional integrity of each, starting at the lowest level  and progressing to the higher levels  of the system until the cause is found . The mos2. The woman exhibited normal secondary sexual characteristics, normal auxiliary and pubic hairs, normal breast development, and agenesis of the upper two-thirds of the vagina, but there was a vaginal pouch without obvious bulging of the hymen. There was no evidence for stress or changes in weight. She was not a professional athlete and never had abnormal eating habits or complains of abdominal pain. Laparoscopy was performed 6 years ago revealing a rudimentary uterus with streak ovaries and a vaginal pouch, whereas the cervix did not exist (β-hCG) concentration was 289 IU/mL on day 12 after insemination, and twin intrauterine pregnancy with fetal heart beat was observed by transvaginal ultrasonography in sixth week of gestation. The patient was followed up by telephone and had cesarean delivery at 36th week of gestation. Now, she has two healthy babies.A 28-year-old nulligravid female presented to our infertility clinic with an 8-year history of primary infertility, sexual dysfunction, and primary amenorrhea. Her body mass index (BMI) was 26 Kg/mot exist . The patTypically, distal occlusions of female genital tract are associated with hematocolpos or hematometra which are manifested with cyclic pelvic pain and amenorrhea . This isEvaluation of primary amenorrhea begins with a careful history and physical examination including the assessment of the internal and external genitalia as well as determination of FSH, thyroid stimulating hormone (TSH), and prolactin concentrations. This approach will identify the most common causes of amenorrhea . PrimaryPatients with AIS have a normal male karyotype  and asymmetrical secondary sexual development (breast development with absent pubic hair), no visible cervix, and a short vagina . TransveHH is manifested by primary or secondary amenorrhea and normal or low serum gonadotropins concentration . HH is aIn conclusion, clinicians should be aware of presence of more than one etiology which causes atypical presentations and accomplishes a systematic strategy for the evaluation of amenorrhea potential to avoid long-term side effects of a misdiagnosis.


Background. Calcified thrombi are a common finding, especially in the pelvic veins. There are generally multiple thrombi, and they are generally associated with vascular malformations. Design. Herein we report a rare case of a single labial phlebolith, not associated with any other vascular lesion. We aim to alert clinicians to the possibility of the occurrence of vascular thrombi in the mouth and to describe the clinical and histological characteristics of such lesions in order to simplify the diagnosis and treatment. Furthermore, we have reviewed the English-language literature published since 1970 reporting oral (including masticatory muscles) phleboliths. Results. Twenty-nine cases of phleboliths have been reported in the literature since 1970. Only three of the reported phleboliths were solitary and not associated with other vascular lesions, as in the case presented here. Conclusion. Although phleboliths not associated with other vascular lesions are not common, clinicians should be aware of the existence of this pathology and include it as differential diagnosis of oral lesions. Phleboliths are calcified thrombi usually associated with other vascular lesions, as vascular malformations (VM), and are caused by blood stasis  or even In the literature, they are commonly found on routine radiographs of cases diagnosed as VM or hemangiomas, although they are better detected by computerized tomography , 3, 4. RThe pelvic area is the most common area affected by phleboliths, which can occur within prostatic, uterine, or intestine veins. Phleboliths are not unusual in the head and neck, but there are few cases in the literature of phleboliths not associated with other vascular anomalies –10. WhenBesides imaging exams, biopsy followed by microscopic examination can help in the final diagnosis. Histologically, a phlebolith is seen as concentric calcifications with an onion-like appearance inside a vessel, caused by repetitive mineral deposition on a thrombus and secondary lamellar fibrosis , 7, 13. We report herein an unusual case of a phlebolith presenting as a single lesion, not associated with a VM and clinically misdiagnosed as a neurofibroma. In describing this case, we aim to help clinicians to deal with this uncommon but simple lesion. We have also reviewed the literature of oral/masticatory muscle phleboliths.A 56-year-old woman was referred to our institution presenting with a white-yellow painless papule in the right labial commissure. It was approximately 3 mm in diameter and firm to palpation and had been growing slowly for approximately one year, following trauma. The patient reported swelling after trauma with subsequent diminution and then a recent increase in volume. Her previous medical history was unremarkable.No radiograph was performed, and the clinical diagnostic hypothesis was neurofibroma and pleomorphic adenoma. An excisional biopsy was performed followed by histopathological examination.Microscopic examination revealed a submucous dilated vessel filled by a calcified thrombus with a concentric pattern of mineralization and surrounded by granulation tissue and small capillaries under a hyaline layer. The covering mucosa was intact and presented no alterations .The diagnosis of phlebolith was confirmed by histochemical Weigert's and Masson's Trichrome staining and by immunohistochemistry with anti-CD34 and anti-smooth muscle actin (SMA) , as evidComplementary electron microscopy with energy dispersive X-ray spectrometry (EDS) revealed the presence of calcium (Ca), carbon (C), silicon (Si), and magnesium (Mg) , compatiMedline and Web of Science databases were searched using the descriptors “oral phlebolith” and “phleboliths,” “phlebolith,” “oral thrombus [case report],” and “lip and thrombus.” We found 29 cases of phleboliths. In five cases, there were multiple phleboliths not associated with other vascular lesions (16.13%), and, in four cases (12.90%), there was a solitary phlebolith, but only one was associated with a lesion diagnosed as hemangioma. Only three reported phleboliths were solitary and not associated with other vascular lesions, as in the case presented here.Based on the reviewed literature, phleboliths of the oral region can be found in infants to elderly individuals, but mainly between the first and third decades of life (55.2%), with no sex predilection , and are sometimes associated with masticatory muscles (27.6%).Phleboliths are calcified thrombi that commonly appear as multiple calcifications related to lesions diagnosed as VM or hemangiomas , 11–21. The diagnosis of a phlebolith is commonly made by physical and imaging examinations and confirmed by microscopy (HE stain). In some cases, immunohistochemistry or even electron microscopy , 14, 15 The EDS showed peaks of C, Ca , Mg, and Si. A study of arterial biomineralization in autopsies found both apatitic and nonapatitic calcifications in different zones of the arterial wall . The litCases of phleboliths not associated with other vascular lesions are rarely reported in the literature, especially as solitary nodules –10. TrauDespite the rarity of this condition, clinicians should be aware of the existence of single phleboliths not associated with other vascular lesions when facing a submucous lesion on the lip or other regions susceptible to trauma and should include them in the differential diagnosis.


Chondrosarcomas are cartilage-matrix-forming tumors that make up 20-27% of primary malignant bone tumors and are the third most common primary bone malignancy after multiple myelomas and osteosarcomas. Radiographic assessment of this condition includes plain radiography, computed tomography, and magnetic resonance imaging for tumor characterization and delineation of intraosseous and extraosseous involvement. Most chondrosarcomas are refractory to chemotherapy and radiation therapy; therefore, wide en bloc surgical excision offers the best chance for cure. Chondrosarcomas frequently affect the pelvis and upper and lower extremities. In rare instances, the chest wall can be involved, with chondrosarcomas occurring in the ribs, sternum, anterior costosternal junction, and posterior costotransverse junction. In this article, we present a patient with thoracic chondrosarcoma centered at the left T7 costotransverse joint with effacement of the left T7-T8 neuroforamen. We also detail our operative technique of wide en bloc chondrosarcoma excision and review current literature on this topic. Chondrosarcomas account for 20-27% of primary malignant bone tumors and commonly affect the pelvis and upper and lower extremities . They arAlthough chondrosarcomas are better known for their involvement of the pelvis and extremities, they are the most common malignant tumor of the chest wall . ThoraciIn this article, we present a patient with thoracic chondrosarcoma centered at the left T7 costotransverse joint with effacement of the left T7-T8 neuroforamen. We also detail our operative technique of wide en bloc chondrosarcoma excision and review current literature on this topic. Informed consent was obtained from the patient for this case report.A 45-year-old right-handed male with no known history of cancer presented to our institution with left-sided flank and abdominal pain that persisted for three months. The patient reported concomitant thoracic back pain with numbness and pruritus in the left T7 dermatome but denied experiencing any other symptom of radiculomyelopathy. A physical examination revealed that he had numbness and hyperesthesia in the left T7 dermatomal distribution. A motor exam revealed that the motor system was intact, and the patient had negative long tract signs.A preoperative unenhanced CT imaging showed a polylobulated mass centered at the left T7 costotransverse joint, measuring approximately 4.8 x 3.3 cm. There was evidence of localized bone destruction as well as an intralesional chondroid matrix. The lesion effaced the left T7-T8 neuroforamen. A preoperative CT scan with coronal Figure  and sagiA preoperative MRI showed a polylobulated, partially calcified, T2 hyperintense, minimally peripherally enhancing mass centered at the left costovertebral joint, at the level of T7, with extension within the left neuroforamina at T7-T8 and T8-T9 . They evaluated 96 patients with primary chondrosarcoma of the chest wall with a median age of 53.5 years. The location of the chondrosarcomas was as follows: ribs (n=78) and sternum (n=18). Of those, 72 patients underwent treatment at the Mayo Clinic; 28 had wide resection, 25 had local excision, and 19 had palliative excision. Local recurrence occurred in 37 patients  and of those, 14 patients developed metastatic disease . In 10 years, recurrence had developed in 50% of patients who had local excision, compared to 17% of patients who had wide resection. Ten-year survival for patients treated with wide resection was 96%, with local excision 65%, and with palliative excision 14%. The authors concluded that survival was influenced by tumor grade, tumor diameter, tumor location, and date of operation .Widhe, et al. (2009) of the Scandinavian Sarcoma Group studied 106 consecutive patients with chest wall chondrosarcomas with a median of nine years follow-up. The patients who had wide surgical resections had a 10-year survival of 92% compared with 47% for those who had intralesional resections. Ten-year survival was also higher in patients treated at sarcoma centers compared to those treated elsewhere (75% versus 59%). As expected, the local recurrence rate was significantly lower when wide resections were achieved (4%) versus intralesional resections (73%). The authors observed that prognostic factors for local recurrence included the surgical margin and histological grade, while prognostic factors for metastases included the histologic grade, tumor size, and local recurrence .Costovertebral chondrosarcomas present an additional challenge to surgical resection. Clinically, extension to the thoracic spine can cause radiculopathy or myelopathy from nerve root or spinal cord compression. In most cases, wide en block surgical resection of chondrosarcomas at the costovertebral joint would require partial or total spondylectomy. Few cases of costovertebral chondrosarcomas have been reported in the literature -15. For Chondrosarcomas are the third most common primary bone malignancy after multiple myelomas and osteosarcomas and is the leading malignant tumor of the chest wall. Thoracic chondrosarcomas usually arise from the ribs , ribs and sternum , or sternum alone ; only rarely do they occur at the posterior costotransverse junction . Costovertebral chondrosarcomas can invade nearby nerve roots and the spinal cord, causing radiculomyelopathy symptoms. They present a unique challenge surgically, and collaboration between thoracic surgery and spine surgery is necessary to achieve wide surgical margins followed by reconstruction and stabilization of the thoracic spine.


Multilobular tumor of bone (MTB)  is an uncommon bone tumor frequently located on the skull of dogs, rarely on the ribs or pelvis. These neoplasms are slow growing, locally invasive, and have the potential to compress and invade the brain. A 10-year-old mixed breed dog was presented with a history of approximately 4 months of progressive growth of a left zygomatic mass. Radiographic investigation revealed a finely granular or stippled non homogeneous radiopaque mass involving the zygomatic arch. After surgery, grossly the neoplasm consisted of multiple, variably sized, grayish-white to yellow nodules separated by collagenous septa of different thickness. Histologically, the tumor was characterized by the presence of multiple lobules containing osteoid and cartilage, separated by a net of fibrous septae. This neoplastic pattern was consistent with a typical multilobular tumor of bone and based on clinical, radiographical, gross and light microscopic findings the definitive diagnosis was made. While reviewing veterinary literature only few cases of MTB were found in dogs. It is also called with alternative terms like chondroma rodens, cartilage analogue of fibromatosis, calcifying aponeurotic fibroma, juvenile aponeurotic fibroma, multilobular chondroma or osteoma and multilobular osteochondrosarcoma . This tumor often recurs locally after surgical excision, and it has been found to metastatize the lungs . The tumor is usually present as a firm immovable mass on the surface of skull bones. Direct extension into adjacent structures is common . Depending of the location, the tumor can manifest in various clinical signs in the affected dogs; which include difficulty in mastication, obstruction of sinuses, neurological signs, exophthalmia and disfiguration of the face and head due to the protruding tumor mass .In dogs it is uncommon and primarily represents a disease of middle-aged to older animals, occurring most often in medium or large breeds and rarely in giant breeds .The literature regarding MTB is limited to a relatively small number of cases reported in dogs  and partially thromboplastin time (PTT).All results were within reference limits similar to the values on urinalysis on voided urine. The dog was sedated by 5 µgm/Kg bodyweight (BW) of Medetomidine  and 0,1 mg/Kg BW of Butorphanol  premedication. The induction was achieved using propofol at 4 mg/Kg BW (17 ml EV) followed by isoflurane for maintenance. The surgery was performed using a lateral access, on the left side of dog’s face. After a careful isolation of anatomic structures and the exposure of the tumor, the mass was removed. Grossly the mass consisted of multiple, variably sized, grayish-white to yellow nodules which occasionally contained whitish, hard areas on cut surface interpreted to be mineral.The resected mass was fixed in 10% neutral buffered 10% formalin and submitted to the Dipartimento di Scienze Biopatologiche e Igiene delle Produzioni Animali e Alimentari, Università degli Studi di Perugia, Italy.Histologically, the tumor was characterized by the presence of multiple lobules of irregularly shaped and sized islands and nests containing well differentiated osteoid and cartilage separated by diffuse anastomosed fibrovascular septa .In some areas there were broad poorly differentiated areas of mesenchymal tissue which blended into the lobules and islands of cartilagineous, osseous or osteocartilagineous tissue. Within the islands there were also diffuse areas of ossification and mineralization. The mitotic index of neoplastic cells was always low grade in all the examined samples. According to the characteristics and histological appearance of the tumor a diagnosis of MTB was made.A radiation therapy was declined by the owner and after 3 months a recurrence was not observed and the dog did not present clinical signs.MTBs are slow growing, locally invasive and can compress and also invade the brain. They metastatize to the lungs late in the clinical course and the metastases are frequently small and clinically silent .et al., 2007). Local recurrence is relatively common following surgical excision and is dependent on completeness of surgical resection. Aggressive surgical excision with wide margins remains the treatment of choice and can result in long-term disease remission .Surgical removal is often difficult because of the location of these tumors and local recurrence occurs in about 50% of the cases (Jubb


Papillary-cystic variant of acinic cell carcinoma is a rare tumour confined to salivary gland tissue. Here, we report the first case ever to manifest in a tumour affecting the lacrimal gland, a completely different tissue type, and review the current published literature on this type of tumour. Papillary-cystic variant is a very rare form of acinic cell carcinoma. The handful of cases reported in the literature have manifested mainly in salivary gland tissue, affecting both the major and minor salivary glands. The tumour type behaves in a similar manner to other varieties of acinic cell carcinoma by presenting as a slowly enlarging mass within normal tissue. In this paper, we describe a manifestation of papillary-cystic variant of acinic cell carcinoma (PCV-ACC) presenting as a mass within the right lacrimal gland and discuss the management and arrangements for patient review. This is one of only a few tumours presenting in the lacrimal gland.A 78-year-old lady presented to our service in February 2013 with a 12-month history of a mass over the right eye. The mass had been gradually enlarging but was not associated with any pain or change in periocular sensation. She denied any vision loss, double vision, epiphora, or xerophthalmia. The patient suffered from hypertrophic cardiomyopathy with an implantable defibrillator inserted to maintain cardiac function but was otherwise fit and well.On examination, a firm mass was arising from the palpebral lobe of the right lacrimal gland, underneath the supraorbital rim. Visual field testing and ophthalmoscopy examination were normal. Visual acuity testing using a Snellen chart revealed 20/70 vision in both eyes, which was in a normal range for a person of this age. Cranial nerve, ear, nose, and throat examinations were all normal. A computed tomography (CT) scan reported that the mass was a soft tissue tumour arising from the palpebral lobe of the lacrimal gland but that the orbits and globes were otherwise normal with no tissue compression or invasion .The entire right lacrimal gland was removed in our hospital under general anaesthetic through an orbitotomy. The expectation was that the tumour would be a pleomorphic adenoma and that removal would prevent future carcinoma development requiring more aggressive surgery with loss of vision. Following surgery the patient made a good recovery and was discharged from hospital the day after surgery. Hypromellose 0.3% eye drops were provided for the right eye to compensate for the reduced lacrimation that would now affect that eye.Histopathological assessment of the removed gland revealed that the soft tissue mass within the lacrimal gland was a PCV-ACC Figures , and 4. The patient's case was discussed at our local head and neck cancer multidisciplinary team meeting. When the histological subtype was considered, the meeting advised that a chest X-ray and ultrasound scan of the liver should be performed to assess pulmonary or liver metastases, respectively. Both these investigations were normal and the patient remains under review by our service. No further complications of surgery developed and to date no recurrence or new tumour has developed.Despite being histologically similar, PCV-ACC principally presents in the salivary glands . PCV-ACCSheyn et al. suggested that PCV-ACC presents in the elderly, though Shet et al. later reported a series of exclusively young patients with the tumour , 7. No aIn conclusion, although PCV-ACC is an extremely rare carcinoma, it should be considered in the differential diagnosis of lacrimal gland tumours because the additional investigations required are so different from that provided for the common lacrimal gland carcinomas. Additionally, head and neck surgeons should be aware of the presence and behaviour of PCV-ACC in salivary glands.


Lipoma is a benign tumor infrequent in the oral cavity, particularly in the tongue: indeed, lipomas only represent approximately 0.3% of all tongue neoplasia. Compared to conventional lipoma, fibrolipoma of the tongue is a very rare lesion that accounts for around 25–40% of tongue lipomas, and until now, to the best of our knowledge, only 14 cases have been described in which histological diagnosis of fibrolipoma was specifically confirmed. We report the case of a patient with a voluminous fibrolipoma of the tongue, treated by means of surgical excision. Fibrolipoma excision, like that described in this report, sometimes may be laborious, because fibrous bands appear to be focally infiltrating adjacent tissues, giving rise to some doubts about the nature of the lesion.We report the case of a voluminous fibrolipoma of the tongue in a 71-year-old Caucasian woman.Because of its histological characteristics, abundance of connective and secondary changes/atrophy, fibrolipoma may appear as infiltrating adjacent tissues and may cause doubts of differential diagnosis with malignant infiltrating lesions. Surgical excision is the elective treatment. However, an accurate differential diagnosis, postsurgical histological examination and careful follow-up are required. Lipoma is a benign tumor most frequently found in almost all anatomical sites that have adipose tissue in their structure.However, its presence is relatively infrequent in the oral cavity. The tongue in particular, totally lacking in adipose tissue, is a rare location for a lipoma; indeed lipomas only represent approximately 0.3% of all tongue neoplasia. According to a histological criterion, the World Health Organization (WHO)  classifiA 71-year-old Caucasian woman presented to our hospital with a painless swelling on the ventral surface of her tongue  was undertaken.A contrast-enhanced CT scan showed an oval-shaped neoplasm, with distinct margins, and a rather patchy density Fig. . NonetheOn macroscopic examination Fig. , the masOn histological examination, the lesion showed clear characteristics of mature adipose tissue, without atypical aspects, subdivided by fibrous shoots in multiple adipose vacuums Fig. ; peripheThe postoperative period was uneventful and our patient was discharged a few days later, with her tongue functions perfectly safeguarded.Lipomas are the most common benign neoformation in almost all anatomical sites that present adipose tissue in their structure. They represent 13–20% .In particular, tongue lipomas are extremely rare and represent 0.3% of all neoplasms of the tongue .On the basis of the results of research conducted on the PubMed database (performed by searching for the keywords ‘tongue’ and ‘lipoma’), since 1912, 185 cases of lipoma of the tongue have been reported and 14 cases of tongue fibrolipoma . Tongue They are generally localized under the mucous membrane. However, Colella et al.  have desAccording to a histological criterion, the WHO  classifiCompared to conventional lipoma, other histological variants are much more rare, and fibrolipoma is particularly rare . Until nThey vary in dimensions, and lipomas of considerable size have been described , 3. The Histologically, fibrolipoma is composed of mature fat cells subdivided into lobules by fibrous shoots. Fibrous connective tissue, especially peripherally, is well represented. From a connective fibrous capsule originate multiple fibrous bands, that often adhere tenaciously to adjacent tissues and structures, with focally pseudo-infiltrating aspects that may cause doubt of a differential diagnosis with malignant infiltrating lesions.It generally presents an ovalar shape with variable volume and dimensions, with a yellow color, and the consistency is generally soft or semi-firm . When coThe etiopathogenesis of lipomas and fibrolipomas is still unknown, even though an alteration of the lipidic metabolism or an anomalous localization of fatty-fetal tissue in the tongue have been suggested. It is thought that repeated mild trauma may trigger fatty tissue proliferation , as suggDiagnosis is made by an accurate anamnesis, which can reveal a relation with lipidic metabolism alterations, and a good clinical examination.Clinically, tongue fibrolipomas are generally asymptomatic, not painful, although, because of the space they occupy, they can cause chewing, swallowing or phonation dysfunction. As noted by Manjunatha et al. , it is iAdding to the clinical data or when differential diagnosis is more complex, imaging techniques can also be used, such as a CT scan, together with diagnostic fine needle aspiration cytology (FNAC), which can be helpful to discriminate the nature of the mass. The CT features of fibrolipoma show an ovalar mass with definite margins, and inhomogeneous density (compatible with adipose tissue). Although cytology can guide us, in many cases it is not sufficient to confirm the diagnosis of fibrolipoma absolutely.In fact, because of its pseudo-infiltrating aspects and its tenacious adherence to surrounding structures, histological examination is mandatory to clarify the nature of the neoformation and resolve all doubts. For its histological characteristics, for its possible secondary changes , for itsDifferential diagnosis has to be performed among different pathologies , 13, 15,Treatment consists of surgical excision, according to the classical technique or by the use of diode laser .According to the literature, fibrolipoma of the tongue is a very rare lesion, which accounts for approximately 25–40% of tongue lipomas , and untDue to the abundance of collagen, connective tissue, fibrous bands, possible ulcerative changes of the coating mucosa , and secWritten informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Primary intraosseous cavernous hemangiomas (PICHs) are benign vascular tumors that may involve any part of the body. PICH occurs more frequently in the spine and less commonly in skull. The earliest description in the English literature was in 1845 by Toynbee, who reported a vascular tumor arising in the confines of the parietal bone. Skull PICHs do not always have typical radiologic features and should always be considered in the differential diagnosis of malignant skull lesions. We now reviewed and analyzed related literatures in detail with reporting a rare case of PICH in the left front bone that was surgically resected. The earliest description of skull PICH was in 1845 by Toynbee. Much to our knowledge, there have only been 93 cases of skull PICH reported previously. The number of relevant literatures each year shows a general tendency to increase over time. A timeline of the related publications is available as Figure Primary intraosseous cavernous hemangiomas (PICHs) is a rare bone tumor accounting for 0.7% to 1.0% of all bone tumors.2 Among the skulls, the frontal bone is the most commonly involved, followed by the parietal bone, temporal bone, and less frequently by the occipital bone. The pathogenesis is still unknown but a history of trauma seems to be related in some case reports.3 Total surgical excision is the treatment of choice and the prognosis after complete excision is excellent and recurrence is usually rare. Herein, we present a rare case of a skull PICH in a 17-year-old girl. The clinical presentation, pathology, differential diagnosis, and treatment of this rare disorder are discussed. Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor of this journal. Because of this, there is no need to conduct special ethic review and the ethical approval is not necessary.PICH is mostly seen in middle age and the male-to-female ratio ranges from 3:1 to 2:1.A 17-year-old girl presented with a swelling on the left forehead, which had been slowly enlarging for 2 year. She denied headache, dizziness, and past trauma history. Other medical history and a review of systems were insignificant. On examination, the diameter of the mass on the left frontal bone was about 6 cm long. In consistency, the lesion was immobile, hard, and not tender. There is no abnormality for routine biochemical tests.Plain cranioaural x-ray demonstrated a radiolucent mass and the CT image showed a 9.3 mm × 18.9 mm × 13.4 mm osteolytic lesion within the diploe on the frontal bone near the orbital roof , while hemangiomas in vertebras are usually the capillary type. PICHs of the cranium are rare benign vascular tumors that account for about 0.2% of all bone tumors and 10% of benign skull tumors.2 The earliest description in the English literature was in 1845 by Toynbee, who reported a vascular tumor arising in the confines of the parietal bone.4 PICH arises from the vessels in the diploic space and supplied by the branches of the external carotid artery, arising in the skull vault. The middle and superficial temporal arteries are the main sources of blood supply. Within the lesion, the capillaries are widely dilated separated by fibrous tissue. The pathogenesis of PICH remains unknown. The cause is considered to be congenital, but this has not yet been proven. Some scholars proposed a hereditary nature for “vascular malformations” in the skull with an autosomal recessive inheritance mode. Others indicated that proliferation and differentiation of the undifferentiated primitive mesenchymal cells induced by a variety of stimuli may be the potential etiology. Trauma may also be an important etiology.5They are predominantly seen in patients in their fourth and fifth decades. Unlike the age predominance, our patient was an adolescent female (17-year old). The male-to-female ratio ranges from 3:1 to 2:1.6 Proptosis and impaired vision may appear if the orbit was invaded. Facial nerve paralysis, twitching of oral commissure, pulsatile tinnitus, and hearing loss may occur if temporal bones are involved. Patients may rarely present with an associated epidural hematoma or subarachnoid hemorrhage. We reviewed some previously reported cases of skull PICH since 2000 .23 Because the imaging findings are not specific, preoperative diagnosis is difficult and histopathology is essential. A thorough clinical, radiographic, pathologic, and treatment comparison to other entities in the differential diagnosis is seen in Table The differential diagnosis for intradiploic skull masses includes dermoid tumors, metastatic diseases, meningiomas, sarcomas, Langerhans cell histiocytosis, eosinophilic granulomas, fibrous dysplasia, giant cell tumors, aneurysmal bone cysts, osteomas, Paget diseases, and so on.6 The bony defect can be reconstructed by virtue of a variety of methods.24 Relapse is uncommon if sufficient safety margins are achieved. Other treatment options include curettage which can be followed by recanalization and irradiation, which may reduce the tumor volume and has demonstrated symptomatic improvement, but has the risk of radiation-induced carcinoma.4 Radiotherapy alone can only can prevent the tumor from growing, but it cannot eradicate the lesions. In keeping with the most widely recommended technique, we opted for a craniectomy with total resection, keeping a 0.5-cm safety margin. We then performed cranioplasty with titanium plate.The treatment of choice for skull PICH is total resection with an adequate normal bone margin to reduce the risk of bleeding.PICHs of the skull are rare benign lesions of vascular origin, showing so semblable medical imaging findings to many other bone lesions that it is hard to differentiate them. Thus, there hemorrhagic features within operative fields and histopathologic examinations remain as the “gold standards” for diagnostics. Total resection with enough uninvolved bone margins must be attempted. PICH's relapse is rare when this surgery is successful.


Acute abdominal pain following laparoscopic live donor nephrectomy (LLDN) might be a diagnostic dilemma, and prompt diagnosis and management is of paramount importance. Herein, we describe a case of acute appendicitis in a 62-year-old kidney donor who presented with acute abdominal pain 16 days following LLDN with features inconsistent with a diagnosis of acute appendicitis. An ultrasound scan suggested strangulated Spigelian hernia unrelated to the operative wound. Exploration of the wound and mini-laparotomy showed no evidence of wound dehiscence or a hernia, but revealed an inflamed appendix wrapped up with omentum. Appendectomy led to complete recovery of the patient. It is imperative to maintain a high index of suspicion for acute appendicitis in this situation to avoid septic complications that might adversely affect the residual renal function and cause negative impact on kidney donation. To the best of our knowledge, this is the first reported case of acute appendicitis following LLDN. Increasing number of laparoscopic live donor nephrectomies (LLDN) are being performed worldwide. LLDN decreases the disincentives for live kidney donation significantly due to reduced post-operative pain and shorter recuperation time . Post-op2) at a pressure between 8 and 12 mm Hg. A routine laparoscopy revealed no abnormalities within the peritoneal cavity. Mobilization of the left colon, ureter, renal vessels and kidney was performed and the blood vessels were divided using ETS-Flex 45 Endo GIA Linear Cutter . The wound was closed in layers with absorbable sutures. The post-operative course was uneventful and he was discharged to home after two days.A 62-year-old Caucasian male underwent transperitoneal left HLLDN, where access to the peritoneal cavity was gained through a 9-cm long infraumbilical transverse incision. The skin and the anterior rectus sheath were divided transversely and the peritoneum was opened vertically in the midline. Two 12-mm ports were inserted over the left upper abdomen. A pneumoperitoneum was maintained by insufflating carbon-dioxide . An ultrasound scan revealed a 16×5 mm hypoechoic lesion protruding into the space between the abdominal muscles at the lateral edge of the rectus abdominis in the right iliac fossa. Doppler sonography indicated blood flow within the mass; however, there were no visible peristalsis (The complete blood count and renal function tests were within normal limits (hemoglobin: 13.2 g/dL, white blood cell count: 8.8×10istalsis . The lesrectus abdominis muscle. No hernia could be detected. On palpation over the intact peritoneum, an ill-defined lump was felt, hence, the peritoneum was opened. There was some turbid fluid around an inflamed appendix, which was wrapped up by the omentum. Appendectomy was carried out in the conventional manner. Histopathologic study confirmed the diagnosis of acute appendicitis. The post-operative period was uneventful. Eight months later, the patient presented with a left inguinal hernia which was treated with a mesh repair.Under general anesthesia, the lesion was explored through a transverse incision over the most tender site marked preoperatively. The muscles were split lateral to the per se has no relationship with LLDN. However, the possibility of strangulation of the appendix through a defect on the wound could be considered, which was not among the operative findings in our case. The other predisposing factors such as fecolith, atherosclerosis, and obstructing lesions at the base of the appendix were absent. Constipation, which is related to mobilization of the colon and side-effects of analgesics, is a common observation following LLDN. This could predispose to acute appendicitis [Complications following LLDN such as injury to the spleen and intestine, bleeding, rhabdomyolysis, chylous ascites and intestinal obstruction from internal hernia have been described previously , 4. Acutndicitis . There wndicitis , 7.In our case, the diagnostic dilemma caused by the atypical clinical presentation following HLLDN remained unresolved until the appendicitis was confirmed at operation. The ultrasound scan had shown a mass in the intramuscular plain containing omentum or extraperitoneal fat, which prompted the diagnosis of a strangulated Spigelian hernia or wound dehiscence. A computed tomography was not carried out deliberately to avoid contrast-induced nephropathy in the solitary kidney. Septic complications related to appendicitis can seriously affect the residual renal function on the remaining kidney after nephrectomy. This case emphasizes the fact that acute appendicitis should be considered in the differential diagnosis of acute abdominal pain in a perfectly healthy live kidney donor after undergoing LLDN even if the presentation may not be typical. A prompt diagnosis and management is of paramount importance as mismanagement can lead to serious consequences and cause negative impact on kidney donation.


Gastrointestinal stromal tumors are a subtype of mesenchymal tumors. In recent years a significant progress was made in their diagnosis and treatment which led to significant improvement of their prognosis. Endoscopy remains one of the main diagnostic methods. In the rare instance of colonoscopy failure, different approaches are available: different endoscope, computed tomography colonography, capsule endoscopy, use of an enteroscope.We present the case of a 75-year-old Caucasian man admitted for abdominal pain, diarrhea and weight loss. Two colonoscopy attempts failed in a different center and a decision to use colon capsule endoscopy was made. This exam revealed a submucosal mass located in the sigmoid colon. Surgery was performed and a local invading gastrointestinal stromal tumor was removed. This is the first image of a colonic gastrointestinal stromal tumor seen on capsule endoscopy.Colon capsule is a useful diagnostic tool in selected patients after colonoscopy failure or contraindication. Gastrointestinal stromal tumors are a subtype of mesenchymal tumors. Even if they represent only 1 % of the digestive tumors, they had reached celebrity in the last years, due to better understanding of their pathogenesis and progress in their diagnosis due to the new and improved imaging tools. Also, a significant progress was made with a targeted molecular treatment which led to significant improvement of their prognosis.The most common locations of GIST’s are the stomach (50–60 %) and small intestine 30–40 %) and only less then 5–10 % develops in colon, rectum and 1 % in esophagus . Similar0–40 % anEndoscopy, echoendoscopy and computed tomography scan are the mean diagnostic methods. We present the case of a patient with a sigmoid colon located GIST, where diagnosis was established using capsule endoscopy after a failed colonoscopy examination.A 75-year-old Caucasian man presented to our department with recurrent left lower quadrant abdominal pain, weight loss and progressive asthenia. The stools were occasional diarrheic, with no visible blood. The symptoms started a few months earlier and they gradually increased. He had no significant medical history.The clinical examination showed a pale patient, with left lower abdominal quadrant pain, no palpable masses, no ascites and no peripheral edema. The blood tests were within normal limits except mild iron deficiency anemia  and an inflammatory syndrome .The patient had been admitted in a different department in the previous month, where an upper endoscopy that found no lesions and two colonoscopies attempts which failed to pass the sigmoid colon were performed. He underwent a barium enema and he was diagnosed with diverticulosis. His symptoms were interpreted as a diverticulitis attack. He received anti-diarrheics, spasmolytics, and a fluoroquinolone with partial and temporary symptom alleviation.He was referred to our unit for a second opinion. Since he had two failures of colonoscopy and since we had an ongoing study using the Pillcam Colon2 videocapsule for the patients unable or unwilling to undergo colonoscopy, we decided to use the Pillcam Colon2 videocapsule from Given Imaging for the colonic exploration. The use of colon capsule endoscopy was not standard care at our institute at that time, but we considered it was the best alternative for our patient. Lately, according to the current European Society of Gastrointestinal Endoscopy capsule endoscopy guidelines, CCE is feasible and safe for visualization of colonic mucosa in patients with incomplete colonoscopy and without stenosis. .After review of his barium irigography, no colonic stenosis was suspected. A complete colonic examination was obtained by capsule and showed multiple diverticula with no signs of inflammation of the surrounding mucosa. However, we noticed a submucosal mass approximately 3 cm in diameter located in the sigmoid colon Fig. A CT scan of the abdomen and the pelvis showed multiple colonic diverticula and a soft tissue mass of 5 cm diameter with a necrotic centre, with increased thickness of the sigmoid wall. No clear demarcation between the mass and the urinary bladder was visible and important densification of the perisigmoidian fat was also noticed. Although a peridiverticular abscess was possible, viewing the CT aspect corroborated with the capsule images, the diagnosis of a tumor with necrotizing center, probably a GIST it was more likely, as this kind of necrosis is quite specific and previously described in GIST’s [The histopathological examination report revealed a sigmoid GIST with a tumor size more than 5 cm, with spindle cells, CD117 positive and a mitotic rate more than 5/50 HPF and since the patient had no significant comorbidities we considered him for the adjuvant therapy. The patient started treatment with imatinib mesylate, with good evolution at 6 months.The clinical features of GIST’s are variable, related to the presence of mass, perforation or obstruction. The presentation can be with abdominal pain or discomfort, acute or chronic bleeding or anorexia . Some paSpecific morphological and immunohistochemical features establish the definite diagnosis of GIST: they are formed by spindle or epithelioid cells which are expressing CD117 and mutations in KIT or PDGFRα . Approximately 80 % also express CD34. Less then 5 % are c-KIT negative .Endoscopy is one of the main methods of diagnosis. Although the rate of colonoscopy completeness is as high as 97 % in expert centers, a variable proportion (4–20 %) of patients will have an incomplete colonoscopy [After an incomplete colonoscopic examination different approaches can be tried: variable stiffness colonoscope, use of gastroscope, single or double balloon enteroscopy . Changing the centre or the endoscopist is an alternative. However, a first failed colonoscopy is more commonly associated with a lower future success rate, particularly when stopped in the sigmoid . RadioloAt the moment four studies address this issue, three using the first generation colon capsule, the other, from our center, presenting the results of the second generation colon capsule. All studies conclude that colon capsule endoscopy can be very useful in the setting of selected patients after colonoscopy failure or contraindication –12. In tComplete surgical resection is the treatment of choice for GIST’s . ImatiniThe colonic GIST’s are rare and a proper evaluation, diagnosis and treatment are needed for the best outcome. In the instance of failing colonoscopy, a useful alternative is colon capsule endoscopy.This is the first case to our knowledge, presenting the capsule image of a colonic GIST. In our patient the capsule endoscopy made the difference, making the correct diagnosis possible and permitting a specific treatment.


A 3-day-old male newborn presented with a severe proptosis of the left eye leading to exposure keratopathy. He underwent debulking of the cyst and biopsy of the tumour and received the pathological diagnosis of epidermoid cyst of orbit. Clinicopathological features of this rare disease are discussed. Several orbital cystic lesions may occur in the childhood –3. CystiEpidermoid cyst  of orbit is a rare benign congenital tumor that causes proptosis in newborns. The incidence of simple epithelial cyst is uncertain. However, this cyst accounted for 8 of the 340 orbital biopsies in children from the Mayo Clinic series [Herein, we described a newborn that was born with severe proptosis on left orbit.A three-day-old male child presented with severe proptosis, which was present at birth, resembling a large mass protruding from left orbit. The child was born full term via elective cesarean section. His 26-year-old mother was healthy with normal antenatal history.At presentation, his ophthalmic examination showed a large tumor in the left orbit. The tumor was protruding the globe anteriorly, preventing the occlusion of the palpebral fissure, and leading to total lagophthalmos, as well as exhibiting conjunctival chemosis and corneal haze . The rigThe mass was nonpulsatile and nonreducible. Cornea was hazy because of exposure keratopathy. The child did not have any other systemic abnormality. The patient was prescribed topical moxifloxacin, nonpreserved artificial tears, and ointment. However, despite the frequent use of medication a corneal ulcer developed in two days. He had an emergent orbital computerized tomography (CT), which demonstrated a cystic lesion filling the left orbit with no apparent intracranial extension. The lesion was surrounding the globe and stretching the extraocular muscles . Thus, fOrbital cysts of the newborn are usually developmental such as dermoid and epidermoid cysts, cystic teratomas, cephaloceles, microphthalmos, and congenital cystic eye , 6. The In conclusion, in a newborn with large orbital mass, a possible malignancy must be kept in mind. Although it is not being observed frequently, benign epidermoid cyst of the orbit is a possible diagnosis in these cases. For diagnostic purposes and preventing exposure keratopathy possibly leading to a decrease in visual functions, an immediate surgery is usually necessary.


Dear SirA full-term newborn male baby was brought with abnormal genitalia. The baby was a product of non-consanguinous marriage and the antenatal check ups and sonography were unremarkable. The boy had passed urine and meconium before arrival. Baby was warm, pink and active. There was no abnormal facies. Limbs and spine were normal. Local examination  revealed a normal phallus with meatus at the tip of glans but it was positioned in the perineum between the scrotum and anus. The hemiscrotums were well developed and contained testis on either side. The anus was patent and its site was normal. Sonography of spine,cranium and urinary tracts showed no pathology. Echocardiogram was also normal.  A pre-penile scrotum or a complete penoscrotal transposition [CPST] is a rare congenital anomaly where the phallus lies between the scrotum and anus. Less than 20 cases of CPST with intact scrotum are reported in the literature [1]. Most of the cases are sporadic. Usually they are associated with other major anomalies of the cardiovascular, gastrointestinal or renal system [2].The embryological explanation of this condition remains elusive. It is postulated that an abnormal positioning of the genital tubercle in relation to the scrotal swellings during the critical fourth to fifth week of gestation could affect the migration of the scrotal swellings [3]. Another school of thought suggests that the phallic tubercle is intrinsically abnormal and affects the corpora’s development which explains the flaccid and hypoplastic penis usually found in this condition [2]. Kain et al proposed that abnormality in the gubernaculum could also result in defective migration of the labio-scrotal folds leading to these kinds of penoscrotal anomalies [4]. The variety of explanations offered clearly summarizes that the complex interactions resulting in this anomaly is yet to be deciphered. As expected for any rare condition a standardized treatment protocol do not exist. It has to be individualized depending on the specific anatomy. Usually an operative correction is offered at 12-18 months of age [5]. Somoza et al had successfully demonstrated a 3-staged correction, 1st stage being done at 18 months of life after topical Testosterone application [1]. Our patient has been discharged with a plan to do a staged repair at around the same age.This case is being projected to add on to the existing literature about this rare scenario and to emphasize that a search for other anomalies must be undertaken before subjecting them to corrective surgeries.Source of Support: NilConflict of Interest: None


IDH mutations have been demonstrated to confer prolonged survival in patients suffering from gliomas, but the mechanisms underlying the improved prognosis are unclear. While some studies have attributed these observations to an enhanced sensitivity to genotoxic therapies, others have postulated that IDH-mutated gliomas exhibit less aggressive intrinsic biological behavior, including the propensity to invade distant sites. Although most gliomas recur local to the site of initial presentation, some tumors demonstrate distant recurrence, the vast majority of which involve the contralateral hemisphere. Trans-tentorial spread has been described once before, in which a supratentorial glioblastoma was reported to recur infratentorially in the cerebellum.We describe a patient who underwent surgical resection, followed by adjuvant radiation and temozolomide of a World Health Organization (WHO) III anaplastic astrocytoma in the right temporal lobe, exhibiting an IDH1 (R132H) mutation. Twenty-two months after surgery, he developed a second lesion, located in the right cerebellum, suspicious for recurrent tumor versus radiation necrosis. A second surgery was performed, and pathology demonstrated recurrent tumor, consistent with IDH1-mutated anaplastic astrocytoma.This is the first example of trans-tentorial spread in an IDH-mutated glioma, suggesting that despite improved survival, IDH mutations may not preclude gliomas from exhibiting the ability to invade distant sites of the brain. Gliomas are malignant tumors arising from astrocytes within the brain and are classified into grades II to IV, based on histopathological criteria set forth by the World Health Organization (WHO). In recent years, mutations in isocitrate dehydrogenase 1 (IDH1) and IDH2 have been shown to characterize low-grade glioma and secondary glioblastomas [It is well known that gliomas frequently recur despite optimal surgery and adjuvant radiation and chemotherapy. Up to 90 % of all recurrences are found within 2–3 cm from the area of initial presentation , while rIn this report, we describe a patient who underwent surgical resection of a right temporal lobe WHO III IDH1-mutated anaplastic astrocytoma, followed by adjuvant radiation and temozolomide. Twenty-two months after surgery, he developed a second lesion in the right cerebellum, for which he underwent total resection. Pathology again demonstrated an IDH-mutated anaplastic astrocytoma, suggesting similar origin from the original tumor. Our patient is the first example of an IDH-mutated glioma exhibiting behavior of trans-tentorial recurrence. As such, IDH mutations, despite association with improved survival, may not preclude the ability of gliomas to recur distantly from the initial site of presentation.A 34-year-old male was incidentally found to have bilateral papilledema on a routine ophthalmological exam. His past medical history was significant for two episodes of generalized tonic-clonic seizures with negative work-up on imaging, for which he was on anti-convulsant medication. To further evaluate his papilledema, he was sent to the emergency department of a nearby hospital for further evaluation where a non-contrast CT showed a 3.7-cm hypo-dense mass in the right temporal lobe associated with 6 mm of leftward midline shift. A brain MRI re-demonstrated a non-enhancing lesion in the right temporal lobe Fig.  with sigAfter surgery, the patient was referred to the neuro-oncology branch of our institution for further management of his condition. Two months after the surgery, the whole tumor bed was radiated over 30 fractions to a total of 60 Gy but given the large volume of the radiation field, concurrent temozolomide was deferred to reduce risk of radiation-induced necrosis. Over the next year, he received 12 cycles of adjuvant temozolomide, which he tolerated well. Serial MRIs demonstrated attenuation of fluid-attenuated inversion recovery (FLAIR) signal abnormality, and clinically, the patient was doing very well and remained free of neurological symptoms.Over the next year and a half, routine surveillance imaging obtained at approximate 6-month intervals showed local disease control and absence of distant recurrence. However, on an MRI taken 22 months after initial surgery, a new enhancing nodule was found in the right cerebellum Fig.  with locGliomas arising within the cerebellum are rare and reportedly make up 0.4–3.4 % of all gliomas, the majority of which present as grade IV glioblastoma . HoweverIt is unclear whether the improved prognosis associated with IDH mutations is due to an enhanced sensitivity to chemo- and radiotherapies or to less aggressive intrinsic biological behaviors. In support of the former possibility, two large-scale studies demonstrated that IDH mutations conferred higher response rates to temozolomide and combination procarbazine, lomustine, and vincristine therapy in low-grade gliomas and oligodendrogliomas, respectively , 9. HoweAlthough debate continues over the underlying mechanism of increased survival observed in IDH-mutated gliomas, our patient demonstrates that IDH-mutated tumors are indeed capable of trans-tentorial spread and may not require WHO IV histopathological characteristics to manifest such invasive features. Regardless, the surgical approach for such diffusely infiltrating gliomas is challenging but continues to involve maximal surgical resection of areas involving contrast enhancement and FLAIR signal abnormalities . For recThis study was approved under protocol 03-N-0164 by the Institutional Review Board of the National Institute of Neurological Disorders and Stroke at the National Institutes of Health. Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient. A copy of the consent form is available for review by the Editor of this journal.


Mediastinal lymphangioma is primarily a benign lesion and the majority of the cases are found incidentally. These lesions account for approximately 1% of all mediastinal tumors. Here we present a giant mediastinal cystic mass in a 35-year-old female who was presented with severe respiratory distress. On the plain chest radiography and CT scan, a massive left pleural effusion with large parasternal and mediastinal lymphadenopathy was seen. Thoracentesis was performed and 400 cm³ of clear fluid was drained from the left hemithorax. However, a subsequent CT scan with contrast and the same technique 40 days later showed a large cystic mass in the mediastinum protruding to the right and left hemi thoraces. The giant cystic mass was resected via right and left anterior thoracotomies. Histopathological examination revealed a diagnosis of lymphangioma. The patient has been alive and without tumor recurrence and has been followed for 2 years.  The mean age group at diagnosis is reported to be around 36 years, with a male to female ratio of 1 to 3.3 Preoperative diagnosis is usually difficult due to non-specific appearance on imaging studies and most of the cases are diagnosed during or after the operation. It is however important to suspect this condition as these lesions appear to have an excellent prognosis after complete resection of the tumor.3-5 Here we present a giant mediastinal lymphangioma with extension into both sides of hemithorax which has not been previously reported in the literature.Mediastinal lymphangioma is a slow growing mass with benign features, and account for 1% of all mediastinal tumors.A 35-year-old woman who presented with severe dyspnea and sweating was admitted to our tertiary referral hospital in March 2009. Her chief complaint was severe worsening dyspnea. Plain chest radiography and CT scan demonstrated huge parasternal lymphadenopathy and a massive left pleural effusion . She undShe was referred to our facility for further work-up. An informed consent was obtained from the patient in order to use the clinical data and imaging studies for research purposes. In our hospital a second contrast-enhanced chest CT scan and the same techniques was performed 40 days after the initial CT scan. A giant cystic mass was found in the mediastinum extending to both hemithoracis measuring (35×25×9 cm). She was subsequently prepared for urgent surgical intervention.In the right anterior thoracotomy, a right cystic mass (10×9×8 cm) was identified protruding to the anterior mediastinum and left hemithorax. Left anterior thoracotomy was also performed for resection of the left sided component of this large cystic mass (20×15×10 cm) extending from the anterior mediastinum to the left hemithorax . There wOn gross pathology, the mass was demonstrated as a brown-gray colored lesion measuring (35×25×9 cm). Gross pathologic examination of specimens showed a membranous appearance with cystic areas filled with clear fluid . The mic4 Benign cystic lymphangioma is usually localized in the neck (75%), axila (20%) or rarely in the mediastinum (1%).6 Mediastinal lymphangioma is clinically asymptomatic in the majority of cases and it is mostly an incidental finding. Giant mediastinal cysts however may produce respiratory symptoms as was present in our patient. Lymphangiomatic lesions are classified into three types: cystic , simple , and cavernous (micro cystic type).4Cystic lesions of the mediastinum are generally classified as a congenital developmental malformation with involvement of the lymphatic tissues.2,4 CT scan provides helpful information in regard to size, density, and site of the cysts.7 The most common finding on CT scan is usually a homogeneous non-enhancing thin walled unilocular mass. They usually appear as low attenuating masses similar to water, although they can have higher attenuation if there is a combination of fluid, solid tissues, or fat. A multi-septal, loculated mass may also be seen sometimes on CT scan or MRI, which can make the diagnosis of lymphangioma particularly challenging.5 MRI is a useful diagnostic tool in preoperative diagnosis and can differentiate different types of lymphangioma.6 At MRI, the lesions present as heterogeneous signal intensities on T1-weighted images and high signal intensity on T2-weighted images.2,8 Siegle and Pilla indicated that at MRI, lymphangioma can have different signal intensities which may make the diagnosis sometimes difficult. Differential diagnosis of mediastinal lymphangioma includes cystic teratoma and cystic thymoma.4On chest radiographs, lymphangiomas appear as well-defined, round, lobular masses.2 Although no explanation has been given for the mechanism by which mediastinal lymphangioma can protrude into hemithoraces, it is probably similar to the way giant thymolipomas are hanging from the anterior mediastinum and eventually protrude to hemithoraces.9Complications of untreated lymphangioma include infection, chylothorax and chylopericardium. In addition, unilateral or bilateral pleural effusions, which are often chylous, may be present.6 They indicated that, but not similar to the size of the giant mediastinal lymphangioma in this patient. The report of lymphangioma of this size has not been previously reported in the literature. This case shows the importance of timely diagnosis and the imaging findings that can be misleading in the initial encounters, but adequate follow up can reveal the accurate diagnosis. A high index of clinical suspicion may be generated by relevant imaging findings and rule out of other etiologies.Bossert et al. briefly reported a large mediastinal lymphangioma with similar clinical presentation and with protrusion into left mediastinum.7,10 After complete resection, the prognosis of this disease appears to be good.10 Nevertheless, complete surgical resection of lymphangiomas needs to be followed up by imaging to detect any recurrence.2 In conclusion, mediastinal lymphangioma may present as a huge cystic lesion with mass effect but proper diagnosis and surgical management is often followed by favorable prognosis.Surgical management is the treatment of choice, although surgery can become at times challenging due to the mass being adherent to large vessels of mediastinum.


Microcystic/reticular schwannoma is a recently described, rare, distinctive histological variant of schwannoma with a predilection for the gastrointestinal tract (GIT). The authors experienced the first case of a microcystic/reticular schwannoma occurring in the esophagus.A 39-year-old male presented for an obstructive sensation during swallowing of several months duration. Endoscopy revealed a bulging mass with intact mucosa at 30 cm from incisors in the esophagus. The mass was excised and gross examination showed it was a well circumscribed, unencapsulated nodule, measuring 3.5×3.2×1.2 cm. On microscopic examination, the tumor showed a vague multinodular appearance with a pushing border and tumor cells arranged in a microcystic and reticular growth pattern with anastomosing and intersecting strands of spindle cells in a myxoid or collagenous/hyalinized stroma. Tumor cells showed diffuse nuclear and cytoplasmic positivity for S100.The authors report the first case of microcystic/reticular schwannoma of the esophagus. Microcystic/reticular schwannoma is a distinctive histological variant of schwannoma with a benign clinical course. However, its histological findings are non-specific and may cause diagnostic difficulties. Awareness of this uncommon neoplasm with distinct histologic features is essential to prevent misdiagnosis. Schwannomas are benign mesenchymal neoplasm and usually arise in the subcutaneous tissues of the distal extremities or in the head and neck region . GastroiA 39-year-old male presented for an obstructive sensation during swallowing of several months duration. During endoscopy, a bulging mass with intact mucosa was observed in the esophagus at 30 cm from incisors Figure . EndoscoSchwannomas account for 2 ~ 7% of mesenchymal GIT neoplasms and the stomach is the most common site . In factTo the best of our knowledge, this is the first report of a microcystic/reticular schwannoma in the esophagus. However, 12 cases of reticular and microcystic schwannoma in the GIT have been reported   with myxoid change, reticular variant of perineurioma, myoepithelial tumor, extraskeletal myxoid chondrosarcoma, low-grade fibromyxoid sarcoma (LGFMS), and ganglioneuroma with abundant myxoid stroma ,2,7-10. or S-100 ,9. GanglWe describe the first case of a microcystic/reticular schwannoma occurring in the esophagus. Microcystic/reticular schwannoma is a distinctive histological variant of schwannoma with a benign clinical course. However, many pathologists and gastroenterologist are unlikely to be familiar with this entity, which could cause diagnostic difficulties. Awareness of this uncommon neoplasm with a distinct histology is essential to prevent misdiagnosis.A copy of the written informed consent provided by the patient prior to publication has been made available for review by the Editor-in-Chief of this journal.


Osteochondromas are the most common benign bone tumors which are mostly seen in the metaphysis of distal femur, proximal tibia, and proximal humerus. As arising from flat bones such as scapula is a rare case, intra-articular osteochondroma is also rare. When the literature is searched it appeared that the scapula and shoulder joint are an uncommon site for osteochondroma. We present a case in which a patient had an osteochondroma placed in shoulder joint and originated from scapula which is a rare situation determined in the literature. Osteochondroma is the most common benign bone tumor , 2 and iWe present a case in which a patient had pain and swelling of the shoulder due to osteochondroma placed in uncommon site in shoulder joint.A 39-year-old man, with no history of trauma, presented to our hospital with pain and swelling in the right shoulder. Pain and limitation of movement had been increasing over a year. Patient, firstly, had been examined in physical therapy department and performed range-of-motion exercises. During this treatment, swelling was added to his complaints. Since pain and swelling developed gradually patient was directed to our department. On the first examination we detected large swelling on the right shoulder and external rotation, flexion, and extension and in particular internal rotation of shoulder was limited. X-ray examination showed that no abnormal finding was noted. MRI revealed the presence of a 2 cm bony mass adjacent to subscapular tendon and close to the glenoid with the typical features of an osteochondroma and extensive effusion in the shoulder joint .Endoscopically assisted surgery was planned. Under general anesthesia, the patient was placed in lateral decubital position. Endoscopic intervention was implemented through the proper portals. First extensive effusion was identified related to synovitis. A 2 cm bony mass was visualized superior to subscapular muscle tendon and close to anterosuperior site of glenoid. Consequently, the tumor was excised through its base by the way of anterior portal . There wAt clinical examination 1 week postoperatively, the patient had dramatic recovery and regained near-full motion of the shoulder, which was almost pain-free.Generally osteochondroma has no symptom, and diagnosis is made incidentally based on posttrauma X-ray films as a bony mass without pain. Osteochondromas may result in symptomatic complaints that include pain due to adjacent soft tissue impingement including bursa formation, vessel and nerve impingement, and painless cosmetic deformity related to the slowly enlarging exophytic mass (pseudowinging scapula). When the tumor is placed by tendon and bursa formation or as in our case intra-articularly it can become symptomatic. Surgical decision can be made in such circumstances. Aside from pain and cosmetic issues, surgical removal may also be indicated due to malignant transformation which is determined to be 1-2% . It has In our case patient had performed range-of-motion exercises but during treatment had no benefit; besides, pain and swelling were getting worse. Osteochondromas may become symptomatic due to pressure on the surrounding soft tissue such as tendon, bursa, vessel, nerve, and the other anatomic structures. Thus, in the present case, osteochondroma caused pain which was placed intra-articularly and near the subscapular muscle tendon. Joint motion exercises increased irritation of the tendon and synovitis after intra-articular irritation statement was also exacerbating the existing pain clinics. Surgical excision as a result of all these is inevitable for our patients. The patient showed dramatic improvement after the surgery was performed for the mass causing the irritation.In the literature, osteochondral lesions were found to be the most common in metaphyseal site of long bone close to physis, distal femur, proximal tibia, and proximal humerus , 12. Alt