Benign notochordal cell tumor is a benign intraosseous lesion, demonstrates characteristic imaging features. The lesion demonstrates low‐signal intensity in T1‐weighted images, high‐signal intensity in T2‐weighted images, and no enhancement with contrast medium in MRI and slight osteosclerosis in CT. If typical imaging findings are identified, biopsy is not necessary. Benign notochordal cell tumor (BNCT) is a benign intraosseous lesion derived from notochordal cells. They are very common lesions, with a reported prevalence of 20% in autopsy findings Here, we report the clinical features of two BNCT cases wherein the diagnoses were confirmed by histopathological investigation following biopsy.A 22‐year‐old male was referred to our university hospital for the investigation of a sacral lesion detected on lumbar spine MRI at a local orthopedic clinic. His primary symptom was lower back pain upon movement. MRI of the sacral spine revealed an intraosseous lesion with clear margins enclosed by the S1 vertebral body. The signal intensities of the lesion were homogeneously low in T1‐weighted images and high in T2‐weighted images, and no enhancement was observed in T1‐weighted images with a gadolinium contrast medium. There were no extraosseous lesions or enlargements Figure . The lesA 42‐year‐old female was referred to our hospital for further investigation of a sacral tumor identified on pelvic MRI at a gynecologic clinic. The patient was asymptomatic and had no abnormal physical findings. MRI revealed that the signal intensities of the tumorous lesion were homogeneously low in T1‐weighted images and high in T2‐weighted images, and no enhancement was observed in fat‐suppression T1‐weighted images with a gadolinium contrast medium (Figure BNCT is a benign intraosseous lesion derived from notochordal cells. BNCT was first reported by Darby et al. The common sites of BNCT are similar to those of chordoma. In rare cases, there are some reports that BNCT coexists with chordoma. Some reports suggested that BNCT may serve as a precursor lesion of chordoma The imaging findings of BNCT reported characteristic as shown in our cases In image findings, there are several diseases that need to be distinguished from BNCT. Table  In MRI, BNCT is a relatively new type of benign tumor that is not yet widely recognized, although it may be commonly encountered because of the increasing availability of spinal MRI scans. When malignant tumor is suspected in image findings, a needle biopsy should be performed without hesitation. However, needle biopsy of the spine is also accompanied by risk of complications. If it can be determined with characteristic image finding as a benign tumor, it will not bear the unnecessary risk of needle biopsy. Our group no longer performs needle biopsy procedures for patients with typical BNCT characteristics after encountering the cases described above. Knowledge of these typical BNCT characteristics is beneficial to avoid unnecessary expense and invasive procedure in such patients.ST: performed conception and design, and wrote the manuscript. KH: wrote parts of manuscript and reviewed the manuscript, and performed patient examination. TA and HK: performed patient examination. SH: performed patient examination and prepared radiological images. EI and HO: reviewed the manuscript.None declared.


Nephroblastoma and neuroblastoma belong to the most common abdominal malignancies in childhood. Similarities in the initial presentation may provide difficulties in distinguishing between these two entities, especially if unusual variations to prevalent patterns of disease manifestation occur. Because of the risk of tumor rupture, European protocols do not require biopsy for diagnosis, which leads to misdiagnosis in some cases.We report on a 4½-year-old girl with a renal tumor displaying radiological and laboratory characteristics supporting the diagnosis of nephroblastoma. Imaging studies showed tumor extension into the inferior vena cava and bilateral lung metastases while urine catecholamines and MIBG-scintigraphy were negative. Preoperative chemotherapy with vincristine, actinomycine D and adriamycin according to the SIOP2001/GPOH protocol for the treatment of nephroblastoma was initiated and followed by surgical tumor resection. Histopathology revealed an undifferentiated tumor with expression of neuronal markers, suggestive of neuroblastoma. MYCN amplification could not be detected. DNA-microarray analysis was performed using Affymetrix genechip human genome U133 plus 2.0 and artificial neural network analysis. Results were confirmed by multiplex RT-PCR.Principal component analysis using 84 genes showed that the patient sample was clearly clustering with neuroblastoma tumors. This was confirmed by hierarchical clustering of the multiplex RT-PCR data. The patient underwent treatment for high-risk neuroblastoma comprising chemotherapy including cisplatin, etoposide, vindesine, dacarbacine, ifosfamide, vincristine, adriamycine and autologous stem cell transplantation followed by maintenance therapy with 13-cis retinoic acid  and is in complete long-term remission.The use of gene expression profiling in an individual patient strongly contributed to clarification in a diagnostic dilemma which finally led to a change of diagnosis from nephroblastoma to neuroblastoma. This case underlines the importance of gene-expression profiling in the correct diagnosis of childhood neoplasms with atypical presentation to ensure that adequate treatment regimens can be applied.The online version of this article (doi:10.1186/s13104-017-2724-4) contains supplementary material, which is available to authorized users. Neuroblastoma and nephroblastoma belong to the most common malignant solid tumors in childhood. In both cases children might present with a distended abdomen. Although there is a considerable overlap of symptoms in the initial presentation such as abdominal distension, loss of appetite, nausea or hypertension, children with nephroblastoma usually are in a better clinical condition and are slightly older with a peak incidence of 2–3 years . A palpa2), actinomycine D (45 µg/m2) and adriamycine (50 mg/m2) was initiated, accompanied by continuous intravenous anticoagulation with unfractionated heparin (100 IU/kg/24 h) with respect to intravasal tumor manifestation. As abdominal MRI after 2 weeks of treatment displayed a constant renal tumor size but an expansion of the IVC thrombus the anticoagulation regime was changed to subcutaneous low-molecular-weight heparin twice daily . After 4 weeks of chemotherapy, MRI scans showed a slight regression of both the primary tumor mass and the IVC thrombus. After completion of 6 weeks of preoperative chemotherapy, the pulmonary metastases and the emboli in the inferior lobe arteries had regressed on chest CT. Radical tumor-nephrectomy, extraction of the caval thrombus and regional staging-lymphadenectomy were performed without major complications. Histopathology revealed clustering small tumor cells, partially arranged in pseudorosettes with focal calcifications, invading the left kidney. Immunohistochemistry was positive for synaptophysin, a specific marker of neuronal and neuroendocrine tumors and VMAT2, expressed in the CNS and sympathetic postganglionic neurons [Fig. 2 ratio is shown  therefore plays a crucial role with regard to the management of further diagnostic procedures, operative treatment and chemotherapy. However, a marginal risk for misdiagnosing these patients on the basis of radiological imaging remains, especially in tumors that are located at the upper pole of the kidneys.A retrospective analysis of 1609 patients registered in German neuroblastoma trials showed that 1.8% had been treated preoperatively as nephroblastomas . ImportaIn our case clear imaging findings indicative of neuroblastoma were absent but tumor expansion to the inferior vena cava was seen. So far, only few patients have been described in the English literature, reflecting the sparsity of this form of neuroblastoma extension –7. In coNext to locoregional lymph nodes the organ most frequently involved by metastatic Wilms’ tumor is the lung. Our patient presented with bilateral pulmonary metastases, putting further weight on the diagnosis of a nephroblastoma. In contrast, although more than 50% of neuroblastoma patients have widespread disease at diagnosis, lung metastases are rather uncommon. In a large cohort of patients with stage IV neuroblastoma from the International Neuroblastoma Risk group database (n = 2808) the incidence of lung metastases at diagnosis was 3.6%. Patients with lung manifestation rather had prognostically relevant LDH elevation and MYCN-amplification compared to the group of stage IV patients without pulmonary metastases  .In more than 90% of neuroblastoma patients either homovanillic acid or vanillylmandelic acid are elevated  and MIBGHistopathology revealed clustering small tumor cells, partially arranged in pseudorosettes with focal calcification present. No definite conjunction with the renal tissue could be seen. Immunohistochemistry in our case stained clearly positive for synaptophysin Fig. a and vesTo accurately discriminate between tumors of similar histological appearance but different origin, biology and clinical behaviour is an ongoing research topic. In the field of SRBCT and the overlap of nephroblastoma and neuroblastoma two recent reports propose immunohistochemical markers, cyclin D1 and PHOX2B, to be reliable for distinction of neuroblastoma from other malignancies , 22. DifIn summary, we present the clinical case of a patient with a renal tumor in whom conventional diagnostic procedures could not reliably differentiate between neuroblastoma and nephroblastoma. We conclude that genetic molecular approaches can be a valuable and precise supplemental tool for making the right diagnosis and instituting correct therapeutic management.Additional file 1: Table S1. ANN diagnosis prediction.Additional file 2: Figure S1. Timeline according to CARE Guidelines for case reports.


We report a rare life-threatening case of spontaneous rupture of renal metastasis from hepatocellular carcinoma (HCC) that was managed by emergent transcatheter arterial embolization (TAE). A 76-year-old woman diagnosed with HCC presented with acute back pain in her right side and was transferred to our hospital. Initial enhanced computed tomography revealed retroperitoneal hemorrhage from the right kidney, which was retrospectively diagnosed as a spontaneous rupture of the metastatic renal tumor from the primary HCC. Detailed examination identified an active retroperitoneal hemorrhage from the lesion and the patient's condition became hemodynamically unstable; hence emergent TAE was performed. The hospitalization period after the TAE was uneventful and sorafenib was subsequently administered. Unfortunately, two months after the TAE, the tumor locally progressed within the retroperitoneal space. Tumors were controlled by repeated TAE as the patient did not want to undergo a nephrectomy. Consequently, she survived for more than one year after emergent TAE, exhibiting low levels of tumor marker. After rupture of the metastatic renal HCC, tumors were expected to progress into the retroperitoneal space, and nephrectomy was the next possible radical treatment to offer the best chance of long-term disease control.  Hepatocellular carcinoma (HCC) is the third-leading cause of cancer-related death worldwide . DespiteA 76-year-old woman presented with acute back pain in her right side and was transferred to Maizuru Medical Center in June 2015. She was diagnosed with HCC, which had arisen from hepatitis C infection, and Child-Pugh Class A cirrhosis. She underwent a central bisegmentectomy of the liver in June 2011, S6 partial liver resection in February 2012, and two partial resections of the lung in November 2012 and August 2013. She was required to undergo follow-up surveillance every three months. As shown in On admission, the laboratory data indicated severe anemia, as shown in On admission, the blood pressure of the patient was 90/64 mmHg, while her heart rate was 109 bpm with a regular rhythm, respiratory rate was 23 breaths/min, and peripheral oxygen saturation was 100%. Because of hemodynamic instability and rapidly progressing anemia (34.2 g/dL on admission and 27.3 g/dL three hours after admission), emergent renal arteriography was performed , and hemIn agreement with the Japanese analysis performed on autopsy specimens from HCC patients, the occurrence of extrahepatic HCC metastasis was retrospectively calculated as 45.6% in lung, 30.7% in lymph node, 17.8% in peritoneum, 14.5% in bone, and 11.1% in adrenal gland , and, ofOne of the life-threatening complications of HCC is spontaneous tumor rupture, and previous literature has reported the considerable mortality rates associated with this . AlthougIn the present case, despite the administration of sorafenib, the tumor progressed into the retroperitoneal space two months after the emergent TAE. Several treatment strategies for intrahepatic HCC have been proposed ; howeverA limitation of the diagnosis of our case was that histology from the right renal tumor was not obtained over the treatment course, and the tumor was not pathologically proven as metastasis from HCC. However, the fact that the TAE for the renal tumor led to normalization of PIVKA-II and AFP levels strongly suggested that the tumor was an extrahepatic HCC lesion . In addiIn summary, we examined a rare case of spontaneous rupture of renal metastasis from HCC, with the resulting retroperitoneal bleeding successfully controlled by emergent TAE. After the rupture of a metastatic renal HCC, tumors would be expected to progress into the retroperitoneal space, as demonstrated in the present case; nephrectomy is the next possible treatment option to offer the best chance of long-term disease control.


Testicular torsion is one of the main causes of acute scrotum and may result in permanent damage of the testicular tissue. Color Doppler imaging has been frequently used in the diagnosis of testicular torsion and posttreatment follow-up period of the disease. There are some studies reporting the value and usefulness of diffusion-weighted imaging in the diagnosis of testicular torsion. However, to the best of our knowledge, there is no report regarding the monitoring of preoperative external detorsion in testicular torsion with diffusion-weighted imaging examination. In this article, diffusion-weighted imaging findings in the management of preoperative external detorsion in a case with testicular torsion were presented. Diffusion-weighted imaging (DWI) is a magnetic resonance imaging (MRI) technique which reflects the movements of water protons. Various pathologies such as tumor infiltration, inflammation, and ischemia affect the diffusion characteristics of the tissue via changing the histological architecture of the tissue, which results in signal differences on DWI. Many studies have reported that these signal changes provide useful contribution in the diagnosis and posttreatment follow-up for various organ and tissue pathologies , 2. To t2, 400 sec/mm2, and 800 sec/mm2 and ADC) technique was performed for the DWI. Twisted right spermatic cord was observed on axial T2-weighted images (A 15-year-old male patient was admitted to our clinic with a complaint of scrotal pain persisting for an hour. Physical examination revealed tenderness and painful right spermatic cord, epididymis, and testicle. There was no sign of scrotal erythema or swelling. The right testicle was located slightly above the oblique position. Laboratory examinations including liver, kidney, and urine analysis were all normal. On ultrasonographic evaluation, there was a heterogeneity and slight increase in the size of the right testicle. Although there was a normal parenchymal blood flow in the left testicle, color Doppler imaging revealed the absence of blood flow in the parenchyma of the right testicle. The patient immediately underwent axial T2-weighted and diffusion-weighted MRI. Magnetic resonance imaging examinations of the patient were performed with a 1.5 T MRI system. A multislice axial plane with a single shot, echo planar spin echo sequence (b: 50 sec/mmd images . The rigd images . The patd images . The patTesticular torsion is one of the causes of acute scrotum, which may result in permanent damage of the testicular tissue. Color Doppler imaging has been frequently used in the diagnosis of testicular torsion and differential diagnosis of other acute scrotal disorders. It has been also reported that it could be used for the evaluation of testicular perfusion after the preoperative external detorsion in acute testicular torsion –5. Howev in vivo. The diffusion of water protons in tissue is affected by several factors including organization and cellular intensity of tissue and structure of extracellular distance [DWI is a noninvasive imaging method reflecting the motion of water protonsdistance , 2, 6. Idistance .Histologically, testicular ischemia has been shown to result in slight interstitial edema and blood congestion in the capillaries causing no changes in cellular morphology , 7. In oIn conclusion, this case report demonstrates that the monitoring of preoperative external detorsion of the spermatic cord with DWI could be a useful and valuable method in patients with acute testicular torsion. However, large sample studies are required to provide precise clarification and conclusions for this issue.


Early diagnosis of pheochromocytoma and its proper management can lessen its mortality and morbidity. This case report describes a 24-year-old pregnant woman with an unusual presentation of pheochromocytoma.An Iranian 24-year-old primigravid woman from Kordistan province was referred to our center with left flank pain at 37 weeks of gestation. She had a history of gestational diabetes mellitus since the 12th week of gestation which was managed by insulin administration. She also had a history of pulsatile bi-temporal headache for 2 years prior to her referral to us. She underwent complete abdominal and pelvic ultrasound imaging for her flank pain. This examination revealed a heterogeneous mass of 119 × 87 × 79 mm above her left kidney, highly suspicious of being an adrenal-originating tumor. Subsequently, we consulted an endocrinologist. She underwent abdominopelvic magnetic resonance imaging and her 24-hour urine metanephrine, normetanephrine, and vanillylmandelic acid were assessed. Finally, the diagnosis of pheochromocytoma was confirmed. She underwent a cesarean section and adrenal mass excision at the 40th week of gestation. This timely diagnosis resulted in her proper management and good maternal and neonatal treatment outcomes.Our patient had pheochromocytoma during pregnancy. She had no complaints about hypertension before or during pregnancy until giving birth to her child; her only symptoms were a vague left flank pain, gestational diabetes, and headaches for the past 2 years. The unusual symptom of flank pain led to timely diagnosis and a good treatment outcome. Pheochromocytoma is a catecholamine-secreting tumor originating from chromaffin cells of medullary region of adrenal gland or sympathetic ganglia. Pheochromocytoma accounts for 0.2 to 0.4% of hypertensive disorders and it may lead to severe or lethal hypertensive crisis , 2. Its During pregnancy pheochromocytoma might not have any specific signs and symptoms. It is less prevalent than other hypertensive disorders during pregnancy. Also specific symptoms such as paroxysmal attacks of sweating, palpitation, and blood pressure crisis are probably lower among pregnant women compared to non-pregnant woman . A literAn Iranian 24-year-old primigravid woman from Kordistan province presented to our hospital with left flank pain. She had had the pain for a few days and had received adequate prenatal care until her referral to us. She was diagnosed as having had gestational diabetes mellitus since the 12th week of gestation (her fasting blood sugar was between 106 and 116 mg/dL). Insulin therapy was started at a dosage of 4 units of neutral protamine Hagedorn at bedtime. All fasting and postprandial blood sugars had been controlled well.She also complained of having had pulsatile bi-temporal headaches for 2 years prior to her referral to us. Her headaches responded well to common analgesics. She also mentioned that she had hypertension of 140/90 mmHg at one of her prenatal visits during the 32nd week of gestation. However, the physician had not diagnosed hypertension by rechecking her blood pressure at that time. Although her gynecologist had assessed proteinuria via 24-hour urine sample, no proteinuria was detected (119 mg/dL). Her liver enzyme, creatinine, and platelet counts were all in normal range.At the 37th week of gestation she had presented to us with left flank pain and no urinary symptoms. We did not detect costovertebral tenderness, fever, hypertension, or tachycardia at the primary examinations. Urine analysis was completely normal, so we did ultrasound imaging of fetus and kidneys. Ultrasound imaging revealed no problems with her kidneys, but we found a solid mass with fine cystic component of 119 × 87 × 79 mm above her kidney, highly suspicious for an adrenal-originating tumor. The fetus had no abnormality at the ultrasound imaging.To evaluate the mass, we consulted with an endocrinologist to definitely diagnose the incidentaloma. Abdominopelvic magnetic resonance imaging without contrast showed a well-defined 100 × 95 mm heterogeneous mass in her left adrenal region above the kidney which contained some cystic areas and showed some restriction foci on diffusion-weighted magnetic resonance imaging . Ten days after the beginning of medical treatment she underwent laparotomy and cesarean section. After induction of anesthesia in the operation room, her blood pressure rose to 240/180 mmHg. Blood pressure fluctuations were managed by intravenously administered nitroprusside. First, a cesarean was done and a boy weighing 3300 grams was born. However, he had secondary apnea after some minutes. This was managed by positive pressure ventilation and he responded well. After repairing uterine incision, endocrine surgeons began tumor resection, which measured approximately 12 × 13 cm in diameter, but no surrounding organ invasion was detected , and even general anesthesia , 16, 17.Surgery is the definite treatment of pheochromocytoma, but there is controversy about the appropriate time. The related factors are gestational age, tumor accessibility, presence or absence of fetal distress, and patient’s response to medical treatment . Some auAlthough pheochromocytoma is rare among hypertensive patients especially during pregnancy, our case demonstrated that early diagnosis and close collaboration of gynecologist, hypertension specialist, endocrinologist, anesthesiologist, and pediatrician can result in good maternal and fetal treatment outcomes. However, delay in diagnosis could result in catastrophic results. In our case the patient had severe paroxysmal hypertension after anesthesia induction.


Adenocarcinoma with osteoblastic metastases is classically seen in prostate, breast, and lung primaries. Less common primary sites include thyroid, kidney, and stomach. We present two cases of primary gastrointestinal adenocarcinoma with metastatic osteoblastic activity from two previously unreported sites. The first case represents an esophageal adenocarcinoma arising in a background of intestinal metaplasia that metastasized with osteoblastic activity to the deltoid muscle. The second case demonstrates a Stage IV sigmoid colon adenocarcinoma with osteoblastic metastases to the liver and lymph nodes. The findings indicate that metastases from various gastrointestinal primary adenocarcinomas can have prominent bone formation. Metastatic adenocarcinoma with osteoblastic activity is classically seen with breast, prostate, and lung carcinomas and less commonly with renal, thyroid, and gastric carcinomas . DespiteA 49-year-old male with a multiple year history of gastroesophageal reflux disease presented with a six-month history of a 50-pound unintentional weight loss with progressive dyspepsia, reflux, and dysphagia of solids and some liquids. Esophagogastroduodenoscopy identified a large obstructing esophageal mass located at 35–40 cm with unremarkable stomach and duodenum. The distal esophageal biopsy showed invasive moderately differentiated esophageal adenocarcinoma in the setting of Barrett esophagus . An esopDuring the initial exam, the patient reported a firm mass overlying his right shoulder. Magnetic Resonance Imaging revealed a 4.7 cm heterogeneous soft tissue mass within the right deltoid muscle lateral to and separate from the humeral shaft . The radSince the patient's initial presentation of dysphagia and soft tissue mass, the patient has undergone multiple rounds of chemotherapy and radiation. He since has developed progressive disease with new metastases to the pancreas, iliacus muscle, and axillary lymph nodes.A 38-year-old previously healthy male presented to an outside hospital with bright red blood per rectum with associated fatigue and anemia. He underwent esophagogastroduodenoscopy and colonoscopy. The colonoscopy showed a sigmoid mass with histology confirming adenocarcinoma. The upper endoscopy was unremarkable. Staging computed tomography scans of the chest, abdomen, and pelvis demonstrated diffuse metastatic disease in the liver, colonic lymph nodes, and cardiophrenic lymph nodes. The liver masses showed significant calcifications on the imaging .At this point, he was transferred to our hospital for further management and initiation of preoperative chemotherapy. He demonstrated radiologic response with a decrease in size of multiple liver lesions. A left hemicolectomy, cholecystectomy, and liver excisions were performed. Gross pathologic examination of the hemicolectomy revealed a 2.3 cm circumferential ulcerated mass at the sigmoid colon that was firm and very difficult to section. Histology confirmed the previously reported invasive colonic adenocarcinoma with metastases to lymph nodes and liver. The primary colonic site , metastaStaging workup revealed Stage IV colon cancer. The patient tolerated the operation and is now postoperative three months with an uncomplicated course. He is doing well and currently on a postoperative chemotherapy regimen.Adenocarcinoma of gastrointestinal origin commonly metastasizes to the lymph nodes, liver, and numerous additional sites. The only gastrointestinal carcinomas documented to develop metastases with osteoblastic activity are from the stomach and occur very rarely. Breast and prostate carcinomas are typically seen to develop metastases that are osteoblastic in nature , 3. OtheAfter an extensive review of the literature of adenocarcinomas producing disseminated extraskeletal osteoblastic metastases, we have not found any cases of carcinomas demonstrating this feature and originating from the esophagus or colon. Primary esophageal and colonic adenocarcinoma has been reported to metastasize to osseous sites with osteoblastic activity , 8. BothIt is interesting to note that special AT-rich sequence-binding protein 2 (SATB2), which is a nuclear matrix protein that regulates osteoblastic differentiation , 10, canIn summary, we presented two cases of metastatic extraskeletal adenocarcinoma with osteoblastic activity. Although commonly seen in breast and prostate and rarely seen in kidney, thyroid, and stomach, our cases are the first to be reported originating from the esophagus and the colon.


Spindle cell hemangioma (SCH) is a unique benign vascular lesion. We present a case of SCH in the upper lip of a 41-year-old woman. A submucosal nodular mass 30 × 20 mm in size was observed in the left upper lip. The mass developed 5 years earlier and enlarged after repeated ethanol injections. The mass was elastic firm, mobile, bluish in color, and well demarcated in magnetic resonance imaging. Under the clinical diagnosis of hemangioma, surgical excision was performed under local anesthesia. Microscopically, the lesion was composed of irregular cavernous spaces and multiple solid cellular areas. Cavernous spaces were filled with a mix of erythrocytes and organizing thrombi. The solid areas showed proliferation of spindle-shaped cells arranged haphazardly or in short interlacing fascicles. Immunohistochemically, most cells strongly reacted with vimentin. CD31, CD34, factor VIII, smooth muscle actin, and Wilms tumor-1 reacted with endothelial cells lining the cavernous spaces. The cells within solid areas consisted of mixed cell population with variable reaction for the markers except for factor VIII. From these findings, the diagnosis of SCH was made. Two years after surgery, no recurrence was noted. A review of SCH in the head and neck region is made. Spindle cell hemangioma (SCH) is a unique vascular lesion, which almost exclusively affects the dermis and subcutaneous tissues of the distal extremities. Perkins and Weiss named a solitary single tumor SCH and multifocal lesions crowded within the same region “spindle cell hemangiomatosis” . More thWe present an additional case in the mucosa of the upper lip of a 47-year-old woman and review of the literature on SCH of the head and neck region.A 41-year-old woman presented with a mass on the left upper lip and difficulty in pronunciation. The mass developed after she bit the upper lip 5 years earlier. The volume of mass was not reduced; however, the patient complained of pain. One year after the development, she visited an otolaryngologist. The mass was diagnosed as mucocele and aspirated. However, only blood was aspirated from this lesion, and the lesion's size was not reduced. Two years after aspiration, the size of mass increased, and she visited a plastic surgeon. The lesion was diagnosed as hemangioma by magnetic resonance imaging (MRI). Ethanol was injected into the lesion twice. Although the lesion was slightly reduced at the first injection, it did not change at the second. She consulted a dental clinic two years after the second injection and then was referred to our department.Oral examination revealed a circumscribed submucosal single nodule, approximately 30 × 20 mm in size in the left upper lip. The overlying mucosa was smooth, with bluish discoloration. On palpation, the nodule was elastic firm and mobile . CervicaThe size of excised the lesion was 30 × 20 mm. The specimen had reddish brown surfaces covered by thin-walled capsule including the mucosa of partial lower lip and inflow blood vessels which were well demarcated. Microscopically, the lesion was a well-circumscribed mass surrounded by fibrous connective tissue and showed a variety of cellularity imparting a lobular architecture in low power Figures . The lesImmunohistochemically, most endothelial cells lining the cavernous spaces, spindle cells within solid areas, and epithelioid cells within both areas strongly reacted with vimentin Figures . The endSCH has been diagnosed as various entities including mucocele, hemangioma, pyogenic granuloma, synovial sarcoma, and enchondroma  1–12]. –12. 1–12Approximately 10% of cases are associated with other developmental anomalies or syndromes, including early-onset varicose veins, lymphedema, Klippel-Trenaunay-Weber syndrome, Maffucci syndrome, epithelioid hemangioendothelioma, and superficial cutaneous lymphatic malformations .SCH is relatively uncommon. Only 12 cases have been reported in the soft tissues of the head and neck region  [1–12]. Histologically, SCH shows variable cellularity imparting a lobular architecture on low power. Large, ectatic, vascular spaces lined by flattened endothelial cells are frequently present. The cavernous spaces may contain calcified thrombi, referred to as phleboliths. Between the dilated vascular spaces, there is proliferation of spindle cells composed of endothelial cells, pericytes, and fibroblasts. The endothelial cells often have focal epithelioid features and show focal cytoplasmic vacuolization. Mitotic activity and atypia are low , 13.SCHs resemble the features of Kaposi's sarcoma, such as male predominance and occasional multifocal growth. But Kaposi's sarcoma rarely contains cavernous vessels with thrombi and phleboliths and lacks epithelioid cells, and their spindle cells react for the endothelial marker CD34. On the other hand, SCH does not present the hyaline globules seen in Kaposi's sarcoma or express human herpes virus 8 latent nuclear antigen-1 .Immunohistochemically, the cells lining the cavernous space and vacuolated epithelioid cells are positive for vimentin, CD31, CD34, and factor-VIII-related antigen, supporting the endothelial nature , 12, 15.α-2b and recombinant interleukin-2 were reported to be successful in inaccessible lesions [Surgical excision is the standard treatment for SCH, with excellent prognosis. Although more than 50% of patients may develop new lesions in the same anatomic region several years after initial excision , these a lesions . On the  lesions  reported lesions . In the SCH is considered an unusual vascular lesion in the oral cavity. Awareness of clinical, histologic, and histochemical features is important for differential diagnosis of the lesion from more aggressive vascular tumors to avoid unnecessary treatment. Although the common size of SCH is less than 20 mm, the size of it is guessed to be increased to more than 30 mm by frequent stimulation. In the diagnosis of SCH in head and neck, it is considered CD 31, CD 34, vimentin, and SMA are beneficial. We suppose the surgical removal of SCH is appropriate treatment with good clinical outcome.


Left hemithyroidectomy was performed. Histopathology showed no signs of invasion, and this is consistent with parathyroid adenoma. Immunostaining was positive for expression of chromogranin A and parathyroid hormone. The patient had no episode of pancreatitis after the operation. In a patient with recurrent episodes of pancreatitis, the possibility of complication with hyperparathyroidism should be considered.We report a case of a 66-year-old woman who developed hyperparathyroidism due to a large intrathyroid parathyroid adenoma with episodes of acute pancreatitis. She had previously been treated for acute pancreatitis twice. Serum calcium was 12.4 mg/dL, and intact parathyroid hormone was 253 pg/dL. Ultrasonography and computed tomography of the neck with contrast enhancement revealed a soft tissue mass (28 mm transverse diameter) within the left lobe of the thyroid.  Primary hyperparathyroidism (PHPT) is a common endocrine disorder characterized by hypercalcemia and excessive secretion of parathyroid hormone (PTH) . PHPT isAcute pancreatitis may be induced by cholelithiasis and alcohol abuse in adults; however, the incidence of pancreatitis in patients with hyperparathyroidism was reported to be only 1.5% [A 66-year-old woman was consulted for evaluation of hypercalcemia. She had been treated for acute pancreatitis twice (3 years and 6 months earlier) and had a long history of hypercalcemia  and calcium 12.7 mg/dL , resp.). Abdominal computed tomography (CT) had shown the presence of multiple renal stones, but not gall stones or pancreatic calcifications (not shown). She usually consumed 350 mL of beer 4 times/week. She had no palpable mass in her neck. Endocrine evaluation was performed according to relevant clinical guidelines, and the patient gave written informed consent for all tests performed. Renal function was within normal. As shown in 99mTc-MIBI) scintigraphy demonstrated focal accumulation of increased radiotracer uptake in the left lobe of the thyroid on both early and delayed images , clearly defined, within the left thyroid . T-scored images .Left hemithyroidectomy was performed due to the clearly defined soft tissue mass within the left thyroid. Histopathology showed no signs of invasion, and this is consistent with parathyroid adenoma. The adenoma was composed mainly of chief cells and oxyphil cells, covered with a fibrous capsule . EvaluatThis is an unusual case of hyperparathyroidism due to a large parathyroid adenoma. This present patient had been treated for acute pancreatitis twice. Pooled clinical data suggest an association between PHPT and pancreatitis , 7. SeruParathyroid adenomas usually measure less than 2 cm and weigh less than 1 g. In parathyroid lesions larger than 2 cm, the differential diagnosis between giant parathyroid adenomas and parathyroid carcinomas would be considered . Parathy99mTc-MIBI has been used for preoperative evaluation of PHPT [The incidence of intrathyroid parathyroid adenoma is rare: true one is 0.7%, and partial one is 1.9% . Imaging of PHPT , as demoHypercalcemia may mediate the development of pancreatitis and our patient had earlier been treated for acute pancreatitis twice. However, during short-term follow-up, she had not experienced any episodes of pancreatitis after surgery. In a patient with recurrent episodes of pancreatitis, the possibility of complication with hyperparathyroidism should be considered.In summary, we report an unusual case of hyperparathyroidism due to a large intrathyroid parathyroid adenoma with episodes of acute pancreatitis.


Intra-abdominal desmoid tumors are commonly associated with genetic syndromes such as familial polyposis coli. Radiological cross imaging studies such as CT and MRI are used in the preoperative work-up of such tumors. Postoperatively, CT and MRI are useful in the assessment of recurrent desmoid tumors. MRI is more accurate in tissue characterization. Where possible, surgical resection remains the standard first-line treatment. For patients where surgery is not possible, or the resection margins are not clear, other forms of treatment are possible. These include hormonal and nonhormonal options. We present a case of a recurrent sporadic intra-abdominal (mesenteric) desmoid tumor. Such an entity is rare with few cases reported in the literature. Treatment options regarding intra-abdominal desmoid tumors will be discussed. Desmoid tumors are deep-seated monoclonal myofibroblastic tumors that account for approximately 0.03% of all neoplasms –3. Desmo47-year-old female presented to our Emergency department in 2012. She complained of progressive intermittent abdominal pain. Clinical exam revealed bilateral pitting edema and an elevated jugular venous pressure, with no history of familial polyposis or colorectal cancer. Laboratory work-up was noncontributory. CT revealed an ill-defined hypoattenuating solid mass in the small bowel mesentery , with slThe mass was resected surgically along with a right hemicolectomy and pathology reported it as a desmoid tumor. Mass was noted to have clear margins on pathology. No additional treatment was given. The patients immediate postoperative course was uneventful.Four years later, the patient returned to the Emergency department with a small bowel obstruction. There was no additional change in medical history. CT was repeated showing a recurrent irregular solid hypoattenuating mass in the mesentery of the right lower quadrant . The masMRI advised for further characterization of the right lower mesenteric mass. MRI  shows thIn the four-month interval between most recent scans the mass had grown significantly from 5 × 4 cm on CT to 8 × 7 cm on MR Figures , resp.. On gross pathology, the mass was noted to be adherent to small bowel and firm. The mass was circumscribed and measured 9.2 × 7.3 × 7.2 cm. Focal areas of hemorrhage were noted near the periphery. The tumor did not invade surrounding fat or bowel . MicroscFollowing surgical resection, no adjuvant therapy was administered. Follow-up with radiological imaging was planned.Desmoid tumors are exceedingly rare. Some identified risk factors include abdominal surgery/trauma, women of childbearing age (desmoid tumor growth is thought to be influenced by estrogen), and familial adenomatous polyposis/Gardner's Syndrome. Common presenting complaints are abdominal distention, pain, vomiting, and intestinal obstruction. However, most patients will present with a painless enlarging mass . Intra-aOn CT, desmoid tumors commonly appear as a well-circumscribed homogenous lesion isodense or hyperdense relative to muscle . The CT The course of mesenteric fibroblastic tumors/desmoid tumors is often unpredictable. They can be indolent and spontaneously regress while others can be very aggressive with rapid growth and mass effect . They doSporadic intra-abdominal tumors occur much less frequently when compared to intra-abdominal desmoid tumors associated with familial polyposis coli. Most sporadic tumors are extra-abdominal in location. Management of intra-abdominal desmoid tumors involves a multidisciplinary team approach. Asymptomatic patients are commonly closely observed for disease progression. Resection remains the first line of treatment. Hormonal and nonhormonal treatments remain an option in patients where resection is not feasible or in those where disease free margins are not achieved. Disease free survival following resection depends on tumor size (>5 cm), extra-abdominal location, margin resection status, and history of recurrence . The rec


Fibrous dysplasia is a benign bone disease first described by Lichtenstein in 1938. It is characterized by progressive replacement of normal bone with fibro-osseous connective tissue. When the disease involves craniofacial skeleton, it results in significant disfigurement and other functional problems. This paper reports a case of large craniofacial fibrous dysplasia involving zygomaticomaxillary complex in a 24-year old male patient. Clinical presentation and imaging characteristics of the pathology is discussed in detail. The disease caused significant facial asymmetry which was satisfactorily managed by surgical recontouring. FD is a rare disorder with reported incidence varying from 1:4000 to 1:10,000. It accounts for approximately 2.5% of all bone lesions and about 7% of all benign bone tumors.2Fibrous dysplasia (FD) is a non-malignant disorder of bone caused by somatic mutations of gene which encodes for the alpha subunit of G protein.3Depending on the number of bones involved, FD is divided into two subtypes- monostotic fibrous dysplasia (MFD) and polyostotic fibrous dysplasia (PFD). As the names indicate MFD is characterized by involvement of a single bone, whereas PFD involves two or more bones. Polyostotic form commonly involves craniofacial bones, proximal femur, and rib. About 3% of PFD is associated with endocrinopathies and skin pigmentation (café au lait spots) and is termed as McCune-Albright syndrome. MFD is most common form and accounts for 80 to 85% cases.3 This paper reports a case of large CFD involving zygomaticomaxillary complex. The clinical presentation, imaging characteristics and management of the pathology are described. FD affecting the craniofacial skeleton although may present as a single lesion, cannot be truly categorized as monostotic because the pathology is not limited to a single bone, and often involve two or more adjacent bones. The term “craniofacial fibrous dysplasia” (CFD) is used to describe such lesions which are confined to contiguous bones of the craniofacial skeleton.A biopsy proven case of fibrous dysplasia of facial skeleton, in a 24 years old man was referred to Oral and Maxillofacial Surgery Clinic, Krishna Hospital, Karad for further evaluation and management. The patient presented with a chief complaint of a painless swelling over left side of face. He first noticed the swelling about twelve years back and it had gradually increased to the present size. The swelling had reportedly not increased in size since last two years. Patient’s medical history was non-contributory. There was no previous history of trauma. On examination, a non-tender, bony hard swelling was noted, involving almost entire left mid-face, extending from midline to the lateral canthus of eye mediolaterally and from infra-orbital rim to the level of upper lip, measuring about 7 cm in diameter . The skiground glass appearance. Surrounding area was normal with no evidence of radiolucent capsule  showed a homogenous radiopaque lesion involving left maxilla. The trabacular pattern was altered, with numerous, irregular, short and immature appearing trabeculae, giving an opacified  capsule . Paranasd normal .Computed tomography (CT) scan showed well-defined multilobulated expansile lesion involving the left zygoma, lateral orbital rim , infra-orbital rim, zygomatic arch, maxillary sinus and superior alveolar ridge, measuring about 50×30×45 mm. Internal structure of the pathology  showed peripheral ground glass appearance, surrounding a central zone of fibrous (woven) bone. Areas of ossification were noted within the lesion . Three dimensional reconstructions of CT scan images showed large bony lesion of left ZMC, producing a contour bulge over maxilla, lateral orbital rim, orbital floor and zygoma . The cliAfter adequate reduction of the mass, to attain best possible facial symmetry, the flap was closed in layers. The patient had uneventful postoperative recovery. Histopathological examination of the excised bone specimen showed loose cellular fibrous stroma composed of haphazardly arranged trabeculae of woven bone giving the characteristic Chinese letter pattern, confirming the diagnosis of fibrous dysplasia . The pat4 Signs and symptoms of CFD vary depending on the location and growth of the lesion. The natural progression of the disease is usually slow indolent mass lesion. Slow growing early lesion may get diagnosed during routine clinical examination or as an incidental finding on head and neck radiographs. Fibrous dysplasia is a rare benign bone disease. When it involves the craniofacial skeleton it causes significant morphological, functional and aesthetic disturbances. Craniofacial fibrous dysplasia (CFD) most commonly involves zygomaticomaxillary complex as seen in present case. The other bones commonly involved include cranial base, maxilla and mandible.5 The present case produced a large painless bony mass over mid-face causing significant facial asymmetry and disfigurement. Visual disturbances, hearing impairment, nasal obstruction, pain, paresthesia, and malocclusion are other possible presenting symptoms of CFD though none of these were noted in our case. The only complaint our patient had was of esthetic disfigurement due to the facial asymmetry.Although rare, rapid growth with cortical bone expansion and displacement of adjacent vital structures maybe seen in young children and pre-pubertal adolescents. Progression of the lesions invariably taper off as the patients approach puberty. Although continued active disease is uncommon in adulthood, they have been reported.6Radiographically the appearance of FD varies from a diffuse radiopaque mass, or a mixed lesion or a radiolucency of the involved bone(s), depending upon the stage and progression of the disease. Sontake et al in a review of fibrous dysplasia involving maxillofacial skeleton stated that ground glass appearance was the most common radiographic presentation of internal structure of FD. CFD involving the maxillary sinus may cause cortical expansion over the external surface. Internal expansion of the lesion within the maxillary sinus leads to a reduction in the volume of sinus cavity, although the shape usually remains unaltered.In the reported case the lesion presented as uniform well-defined homogenous radiopaque mass on plain radiography, giving typical ground glass appearance. Internal structure of the pathology on computed tomography showed peripheral ground glass appearance, surrounding a central zone of fibrous (woven) bone with areas of hyperdensity (ossification). History, clinical presentation and the radiographic features are often adequate to establish diagnosis of FD. 7 The primary goal in management of patients with CFD is to correct the bone deformity so as to restore the facial contour and symmetry. Surgical management of the lesion varies from conservative shaving or osseous contouring to more aggressive resection of the lesion in-toto. Histologically FD shows low to moderately cellular fibrous stroma surrounding irregular, curvilinear trabeculae of woven bone, which is arranged in a pattern commonly referred to as Chinese letter form. The stroma of the lesion has varying amount of collagen, with ratio of fibrous tissue to bone ranging from being totally fibrous to being densely packed with dysplastic trabeculae. The microscopic features cannot differentiate various forms of FD and does not predict the biological behavior of these lesions.It is recommended that all the contouring procedure should be carried out after end of the growth phase (i.e. after adolescence). Patients should be informed about the risk of recurrence and need for surgical recontouring or resection in case of growth of the lesion. Satisfactory bone shaping of the lesion was attained for the present case using extended Weber Ferguson approach. Patient at two years follow-up showed no increase in size of the lesion with aesthetically improved facial symmetry and appearance and was very satisfied with his appearance. Thus this paper highlights the features, both clinical and imaging based, to be borne in mind when handling such patients with facial asymmetry. It highlights the role of surgical options that can be made use of to deal with fibrous dysplasia.The authors declare no conflict of interest.


Keratoacanthoma (KA) is a benign epithelial tumor that typically presents as a firm, cone-shaped, flesh-colored nodule with a central horn-filled crater. KA is considered to be a low-grade variant of squamous cell carcinoma (SCC). We report a rare case of a 72-year-old male who presented with a KA involving the nasal septum, possibly related to ranibizumab use. A flesh-colored lesion on the right anterior nasal septum lesion was visualized on examination. Histologic examination revealed a well-circumscribed, dome-shaped central crater filled with keratin, well-differentiated squamous epithelium with ground-glass cytoplasm with pushing margins, and intraepithelial microabscesses establishing the diagnosis of KA. KA of the nasal septum has only been reported once in the literature. This case is unusual because it normally presents on sun-exposed areas. Additionally, this patient was taking ranibizumab, a vascular endothelial growth factor (VEGF) inhibitor for macular degeneration. Despite ranibizumab not being directly linked to precancerous and cancerous skin lesions, agents in this medication class have been. Although it is difficult to prove associations in this isolated case, the role of ranibizumab causing cutaneous lesions should be further investigated. Keratoacanthoma (KA) was first described by Hutchinson as a “crateriform ulcer of the face” . HoweverA 72-year-old Caucasian male presented to an outpatient practice with an approximate 1-year history of a right nasal lesion. He stated that it was cosmetically bothersome to him and requested evaluation. Associated symptoms included mild nasal obstruction, mild pain at the lesion site, and nasal congestion. Patient denied ulceration, bleeding, and drainage. Past medical history included seasonal (pollen) allergic rhinitis, macular degeneration, glaucoma, hypertension, and hyperlipidemia. Past surgical history included carpal tunnel release and meniscal tear repair. His family history was noncontributory and negative for skin disease. Social history revealed no use of tobacco, alcohol and illicit drugs. Current medications included pitavastatin, losartan, amlodipine, omeprazole, naproxen, and ranibizumab.Vital signs obtained in the office were stable and within normal limits. The physical examination was only significant for a right anterior nasal septum lesion. It was well-circumscribed, flesh-colored, dry, and mildly tender to palpation. There was no ulceration or drainage appreciated. The lesion appeared to be localized with no extension. Cranial nerve examination was unremarkable. No other lesions or lymphadenopathy were noted on examination.The decision was made to bring the patient to the operating room for wide local excision of this lesion. Examination under local anesthesia revealed a well-circumscribed, flesh-colored, crateriform lesion along the anterosuperior portion of the right nasal septum. Full excision of the lesion was performed and the surgical specimen was sent to the pathology laboratory for review. Gross examination revealed a 0.6 × 0.3 cm lesion which contained a centrally located ovoid papule measuring 0.3 × 0.3 × 0.2 cm.Histologic examination of the lesion revealed a well-circumscribed, dome-shaped central crater filled with keratin . A pushiKA is a rapidly growing cutaneous neoplasm most commonly seen on the face, forearms, legs, and hands. It usually affects light-skinned, middle-aged to elderly individuals, with a peak incidence between the ages of 50 to 69 years. Its time course often spans 4 to 6 months from origin to spontaneous involution but some may persist for over one year . KA of tBroadly speaking, the differential for a skin lesion of the nose can be divided into nonmalignant tumors, inflammatory conditions, vascular lesions, premalignant lesions, and malignant tumors. Nonmalignant tumors would include freckles, comedones, adenoma sebaceum, hidrocystoma, fibrous papule, sebaceous hyperplasia, and rhinophyma. Inflammatory conditions affecting the nose include pemphigus, sarcoidosis, systemic lupus erythematosus, facial eosinophilic granuloma, rosacea, herpes zoster, leishmaniasis, and leprosy. Vascular lesions observed in the nose are telangiectasia, hemangioma, and spider nevus. Premalignant lesions involving the nose include actinic keratosis and keratoacanthoma. Finally, malignant tumors of the nose include basal cell carcinoma, squamous cell carcinoma, and melanoma .More specifically, there is also wide differential for lesions of the nasal septum. This includes congenital lesions , granulomatous diseases (Wegener's granulomatosis and sarcoidosis), and neoplasms  as well as infectious conditions  .The origins of KA have not been completely elucidated but there are various causative theories which exist . Its assRanibizumab is a vascular endothelial growth factor (VEGF) inhibitor that has been used for the management of macular degeneration since 2007 . There h


A seeded fistula-in-ano from a synchronous sigmoid colon cancer is rare. The literature is still divided regarding the standard treatment, although an abdominoperineal resection is considered the best option when curative resection is possible.This case is distinct from previous reports, as the patient had known metastatic liver disease before surgery, and proceeded with a pan-proctocolectomy after neo-adjuvant chemotherapy. The patient died 20 months post-operatively of his metastatic liver disease, having been otherwise asymptomatic for eight months on continued chemotherapy, before commencing palliative treatment (completed five cycles).Given its rarity, a low suspicion to biopsy a fistula-in-ano is advocated, and the exclusion of malignancy should be considered prior to surgery. A seeded fistula-in-ano from a synchronous adenocarcinoma of the colon or rectum is rare. Till date, only 28 cases have been reported since the report by Guiss, et al. in 1954 . Given iIt is now widely recognised that exfoliated cancer cells can implant distally on abnormal or damaged mucosa, including wounds and anastomotic or iatrogenic sites; stapled or biopsied -6. The sA 53-year-old man presented with a four-week history of a discharging perianal lump. This had been causing him intermittent discomfort and fresh rectal bleeding. He had a long history of symptoms which had been attributed to haemorrhoids; he was taking irbesartan and atorvastatin tablets for his hypertension and hyperlipidaemia respectively. His sister had Hodgkin’s lymphoma and there was a family history of bowel cancer from his grandmother. He had never smoked but consumed alcohol thrice a week.On examination, his abdomen was soft and non-tender, while his perineum revealed external haemorrhoids and a hard lump (2 cm in diameter) with two discharging points in the right anterior perianal position  which looked concerning. A digital rectal examination revealed what felt like an extra-sphincteric mass. The patient was immediately forewarned of the possibility of a malignant infiltrated fistula tract and a proximal bowel cancer.Initial investigations included a colonoscopy, examination under anaesthetic, and a staging computed tomography (CT) scan of his abdomen and pelvis, which revealed no liver metastases or lymphadenopathy. On colonoscopy, there were two synchronous sigmoid tumours (4 cm and 5 cm in diameter), which were 25 cm and 30 cm respectively from the anal verge. The proximal tumour prevented progression of the scope. Biopsies of both these tumours revealed infiltrating adenocarcinoma.A rectal polyp (6 mm in diameter) was found 6 cm from the anal verge, and the previously mentioned right anterior perianal hard lump was 2 cm from the anal verge. The hard lump had two fistula openings. The Lockhart mummery probes demonstrated that these were high right anterior trans-sphincteric fistula-in-ano’s. Histology of the external openings of the fistulas revealed skin with infiltrating adenocarcinoma, while the rectal polyp showed ulceration with acutely inflamed granulation tissue.The patient was discussed at a multi-disciplinary meeting and subsequently underwent long-course neoadjuvant radiotherapy with 45 Gray (Gy) to the pelvis and perineum, with a 5.4 Gy perineum boost, and chemotherapy with 5-fluorouracil.The patient was planned for a repeat CT scan at six weeks post adjuvant therapy and for surgery four days after. On the day of the CT scan, the patient developed a large bowel obstruction (LBO), but underwent the CT scan which revealed an LBO from the distal sigmoid cancer and multiple 1-2 cm liver metastases. He was admitted that same day and an uncovered stent was placed through the obstructing distal sigmoid cancer. Four days later he underwent a pan-proctocolectomy and end ileostomy formation  scan revealed a ‘mixed response’ to chemotherapy, showing new liver lesions and no extra-hepatic disease. These findings were consistent with a rising carcinoembryonic antigen (CEA) of 1500 µg/L and worsening liver function tests. The patient continued on chemotherapy and was otherwise asymptomatic for eight months. There was initially interval reduction in size and volume of the hepatic metastases. However, the patient’s liver metastatic disease progressed and his treatment changed to folfiri and cetuximab as palliative treatment (completed five cycles). The patient eventually died of his metastatic liver disease 20 months post-operatively.We describe a case where synchronous sigmoid colon cancer seeded a fistula-in-ano. Although rare, other case reports have also concluded this when implanted cells in the fistula share the same histology as the primary cancer from the sigmoid or rectum . This inA small percentage, 0.1% of all anal fistulae, will however represent a primary cancer resulting from a persistent anal fistula, with recurrent inflammation for at least 10 years and in the absence of a proximal primary adenocarcinoma [With only 28 reported cases of an adenocarcinoma seeding a fistula-in-ano, there is still a divide in the literature with regards to a standard treatment -2. An abThis case highlights the rare nature of a synchronous sigmoid colon cancer seeding a fistula-in-ano. The best surgical approach remains controversial. Nevertheless, a low suspicion to biopsy a fistula-in-ano is still advocated, as should the exclusion of proximal colorectal pathology, including malignancy.


Plantar fascia (PF) disorders commonly cause heel pain and disability in the general population. Imaging is often required to confirm diagnosis. This review article aims to provide simple and systematic guidelines for imaging assessment of PF disease, focussing on key findings detectable on plain radiography, ultrasound and magnetic resonance imaging (MRI). Sonographic characteristics of plantar fasciitis include PF thickening, loss of fibrillar structure, perifascial collections, calcifications and hyperaemia on Doppler imaging. Thickening and signal changes in the PF as well as oedema of adjacent soft tissues and bone marrow can be assessed on MRI. Radiographic findings of plantar fasciitis include PF thickening, cortical irregularities and abnormalities in the fat pad located deep below the PF. Plantar fibromatosis appears as well-demarcated, nodular thickenings that are iso-hypoechoic on ultrasound and show low-signal intensity on MRI. PF tears present with partial or complete fibre interruption on both ultrasound and MRI. Imaging description of further PF disorders, including xanthoma, diabetic fascial disease, foreign-body reactions and plantar infections, is detailed in the main text. Ultrasound and MRI should be considered as first- and second-line modalities for assessment of PF disorders, respectively. Indirect findings of PF disease can be ruled out on plain radiography.Teaching PointsPF disorders commonly cause heel pain and disability in the general population.• Imaging is often required to confirm diagnosis or reveal concomitant injuries.• Ultrasound and MRI respectively represent the first- and second-line modalities for diagnosis.• Indirect findings of PF disease can be ruled out on plain radiography.•  Plantar fascia (PF) disorders are common in the adult population . They caThe PF Fig. , also caPlantar fasciitis Figs. , and 4 iPlantar fibromatosis or Ledderhose disease Fig.  is a benTears of the PF Fig.  are uncoPeculiar lesions of the PF should be kept in mind as differential diagnoses of the main PF disorders and include xanthoma, diabetic fascial disease, foreign-body reactions and plantar infections. Xanthomas Fig.  are descPlain radiography is widespread, cost-effective and panoramic, thus often representing the imaging modality of first choice for the evaluation of painful heel.Several findings suggestive of plantar fasciitis can be detected on conventional radiographs. Despite this, plain radiography should not be used to make a diagnosis of plantar fasciitis without knowledge of clinical history or physical examination findings . PF thicPlantar calcaneal spurs and calcifications within the PF are uncommon occurrences in patients with plantar fasciitis , 20. TheApart from highlighting indirect signs of plantar fasciitis and calcaneal spurs, conventional radiography is useful in overviewing anatomical and pathological changes of the bone and soft tissues. Radiopaque foreign material, such as metals, may be easily revealed . In the On ultrasound, similarly to ligaments, PF shows a fibrillar pattern due to the hyperechoic appearance of type I collagen fibre bundles embedded within a background of hypoechoic matrix , 46.Sonographic characteristics of plantar fasciitis include loss of fibrillar structure, increased thickness over 4 mm, perifascial collections and calcifications within the PF Figs.  and 4 1, 47–57. The sonographic presentation of plantar fibromatosis (Ledderhose disease) includes typically single, rarely multiple iso-hypoechoic, well-demarcated, nodular thickenings of the PF, with no calcifications or fluid collection. Doppler ultrasound generally shows no vascular flow inside the lesion Fig.  64]..64].Sonographic findings of PF rupture include complete or partial interruption of the PF, with hypoechoic tissue at the site of rupture related to local haemorrhage and inflammation Fig.  3]..3].Among other disorders of the PF, plantar xanthomas appear as nodules with a speckled pattern . IncreasIn healthy individuals, the PF is homogeneously hypointense on both T1-weighted and fluid-sensitive sequences .MRI findings of plantar fasciitis include: thickening of the PF, most commonly at its calcaneal origin; intrasubstance areas of intermediate signal on T1-weighted sequences and increased signal on fluid-sensitive sequences; oedema in the adjacent soft tissue; bone marrow oedema of the calcaneal attachment of the PF suggestive of enthesopathy Figs.  and 3 22, 4.In plantar fibromatosis (Ledderhose disease), the common MRI appearance of plantar fibroma is a lobulated mass of low signal intensity on both T1- and T2-weighted sequences due to its fibrous nature Fig. . In someMRI findings of acute PF tear are complete or partial interruption of the low signal of the PF and signal changes at the site of lesion including high signal on fluid-sensitive sequences and intermediate signal on T1-weighted sequences. High-signal intensity may be an additional finding on fluid-sensitive sequences in the soft tissues surrounding the site of rupture Fig. ; this reAmong other lesions of the PF, xanthomas appear as fusiform enlargement of the PF showing heterogeneous signal intensity on both T1- and T2-weighted sequences Fig.  66]. In. In66]. Based on a systematic review of the last 20 years’ literature and taking advantage of our centres’ experience, this study is able to shed light on key features of PF disorders that can be identified on conventional imaging modalities, such as plain radiography, ultrasound and MRI, thus representing a valuable guide to proper diagnosis of PF disease. Radiographic, sonographic and MRI findings of PF disorders are summarised in Table Plantar fasciitis is the most common disorder of the PF and a frequent cause of heel pain in the general population . Plain rIn cases of atypical clinical presentation or where imaging findings do not confirm the presence of plantar fasciitis, differential diagnosis includes other causes of PF disease, such as plantar fibromatosis, trauma and infection, but also disorders arising from structures other than the PF. Ultrasound has several advantages over MRI in the assessment of Ledderhose disease or plantar fibromatosis. Plantar fibromas may be small, thus appearing as small hypointense lesions on MRI, and are difficult to differentiate from the low signal intensity of the PF. Small plantar fibromas are more easily detected on ultrasound because of the contrast between their poorly reflective echotexture and fibrillar appearance of the normal PF. Further, both feet may be examined together using ultrasound. Examining both feet together using MRI reduces the in-plane resolution; in contrast, examining both feet separately is time-consuming if compared with ultrasound . A signiSeveral pathologies involving structures other than the PF may mimic PF disease and should be included in the differential diagnosis. Achilles tendinopathy may present with symptoms that are similar to those of plantar fasciitis. This may be related to the close anatomic connection between the PF and the paratenon of the Achilles tendon , 75. ThuIn conclusion, PF disorders are common causes of heel pain and disability in the general population. Imaging is often required to confirm diagnosis or reveal concomitant injuries. As an inexpensive, quick and dynamic imaging technique that also provides high-resolution depiction of the PF and comparison with the contralateral side, ultrasound should be considered the modality of first choice for assessing PF disorders. Several indirect findings of PF disorders can be detected on conventional radiographs and should be identified even in patients examined for other reasons. MRI can reliably delineate both the soft tissue and bone anatomy of the sole of the foot and enables correct diagnosis of PF disorders, but is expensive and should be regarded as a second-line imaging modality.


Extraskeletal primitive neuroectodermal tumor (PNET) is rare and that affecting the female genital tract is extremely rare. We report the case of a 17-year-old girl who presented with swelling of the clitorus. She underwent excision and histopathology was PNET. She received chemotherapy and radiation and achieved complete remission. However, she relapsed locally and systemically soon, progressed and died. Ewing’s sarcoma (ES)/primitive neuroectodermal tumor (PNET) is a high grade malignant neoplasm that mainly involves the skeleton. Primary extraskeletal PNET is rare, comprising 1% of soft tissue sarcomas . We repoA 17-year-old girl presented with swelling at the clitorial region of few weeks duration. On examination, there was a swelling 3 × 2 × 2 cm at the clitorius. Computerized tomography (CT) scan of the abdomen and pelvis showed a right ovarian cyst of 3.5 × 2.5 cm in addition to the clitorial mass. She underwent diagnostic laparoscopy and left ovariectomy for the ovarian lesion and excision of the clitorial mass at the local hospital and was subsequently referred to us. Her performance status was 0, blood and serum chemistries were normal and lactate dehydrogenase was 405 U/L. The histopathological examination of the specimen from clitoris showed a neoplasm composed of round cells in large lobules separated by fibrocollagenous stroma, cells with scanty to moderate cytoplasm and vesicular nuclei, and many thin walled blood vessels were seen . The tumPNET is considered an entity of Ewing’s family of tumors and has a similar natural history, prognosis, immunohistochemical and cytogenetic profiles . They acExtraosseous PNET is also now regarded as a member of the ES/PNET family. It typically involves the soft tissues of the chest wall, pelvis, paravertebral region, abdominal wall, retroperitoneal region and extremities of children, adolescents and young adults . PNETs oPNET of the external genitalia usually presents as a rapidly growing mass, with watery and foul smelling discharge per vagina or with pressure symptoms such as tenesmus and difficulty in passing urine .A 25-year-old girl with sickle cell disease who developed PNET of vagina, treated with surgery, radiotherapy and chemotherapy was alive at 1 year . CetinerEWS gene on chromosome 22 and the FLI-1 gene on chromosome 11. Vaginal PNETs were also found to have EWS and FLI-1 genes by molecular study [PNETs are round cell tumors showing varying degree of neuro-ectodermal differentiation. Immunohistochemical studies help in differentiating PNET from other round cell tumors. Positivity for CD99, a product of MIC2 gene is useful in confirming the diagnosis but it is not specific. Other markers such as vimentin, FLI-1 protein, synaptophysin, neuron-specific enolase and S100 protein are also positive in PNET. Lack of desmin, myogenin and myoD1 can rule out rhabdomyosarcoma and LCA negativity excludes lymphoma . In our ar study .Surgery, chemotherapy and radiotherapy are the treatment for PNET . The cheManagement of these patients involves multidisciplinary approach to improve the outcome and survival rate.


An emergency department encounters a variety of cases, including rare cases of the strangulation of a mesenteric lipoma by the greater omentum band.A 67-year-old Japanese man presented with nausea, vomiting, and upper abdominal pain. There were no abnormalities detected by routine blood tests other than a slight rise in his white cell count. A contrast-enhanced computed tomography scan of his abdomen revealed a dilated intestine, a small intestine volvulus, and a well-capsulated homogeneous mass. He was suspected of having a small intestine volvulus that was affected by a mesenteric lipoma; therefore, single-port laparoscopic surgery was performed. Laparoscopy revealed a small intestine volvulus secondary to the strangulation of a mesenteric lipoma. The band and tumor were removed. He had no postoperative complications and was discharged on postoperative day 6.Although this case was an emergency, it showed that single-port laparoscopic surgery can be a safe, useful, and efficacious procedure. Lipomas are benign neoplasms of adipose tissue that can occur almost anywhere. Mesenteric lipomas are uncommon , but strA 67-year-old Japanese man presented with a 1-day history of nausea, vomiting, and upper abdominal pain. He described that these symptoms had occurred intermittently for several years, and had previously alleviated naturally; however, in this instance, they did not alleviate. Although he used to take medication for hypertension, he had no surgical, family, social, or environmental history. A physical examination revealed only a slightly distended abdomen. A neurological examination showed no abnormality. His vital signs on admission were: blood pressure 152/96 mmHg, heart rate 54 beats per minute (bpm), and body temperature 36.5 °C. A laboratory investigation revealed a white blood cell count of 11,900 cells/μL comprising 82.6 % segmented neutrophils (Fig. en bloc. Surgical blood loss was 20 ml, and operative time was 92 minutes. Our patient had no postoperative complications and was discharged on postoperative day 6. A histopathological examination revealed that the tumor was composed of mature adipocyte-like normal adipose tissue, confirming that it was a lipoma (Fig. He was placed under general anesthesia in the supine position. A 2 cm incision was created in his umbilical region, and covered with a wrapping protector. Three 5 mm laparoscopic cannulae were introduced through the access channels of the multi-trocar device at the 3, 7, and 11 o’clock positions. Following initial abdominal exploration, a twisted small intestine and congested tissue were found. The congested tissue was caused by the greater omentum band Fig. , resectiA lipoma is a benign tumor of mature adipocytes. A lipoma can occur almost anywhere in the trunk, extremities, or even intraperitoneally, which is extremely rare with a small overall malignant potential . The difA lipoma is often accidentally detected secondary to other symptoms. The symptoms in many cases are abdominal swelling, abdominal pain, or mass palpation . The symThe tumor was identified; however, the nature of its involvement in the twisting was not known. Therefore, the first operation was performed to resolve the twisting. Single-port laparoscopic surgery was chosen because the intestinal volvulus was believed to be corrected and it was expected that the surgery would not be very challenging to perform. In the case of the cancellation of the volvulus, the enucleation of the tumor, and the partial resection of the small bowel, laparoscopic surgery, specifically single port, is a very good adaptation. In the case of a small open surgery, we may overlook a tumor that is in other locations because we cannot observe it properly. In addition, pain reduction and early resumption of oral food intake are more likely to lead to shortening of the hospital stay. However, it is preferable for an operation to be performed by an expert surgeon because it is necessary to resolve the twisting and observe the entire small intestine at which time complications such as intestinal tract damage may be caused by the operation of forceps. If the operation is difficult, the addition of a port or a small laparotomy should be performed. We must remember that safety is the topmost priority.The strangulation of a mesenteric lipoma by the greater omentum band is rare. To the best of our knowledge, this is the first report of not only the pathophysiology but also the treatment by single-port laparoscopic surgery.


The extra-adrenal paraganglioma is a neoplasm originating in regional structures, uncommon in paediatrics. We report on a case of a 13-year-old patient who began with severe arterial hypertension, tachycardia, dilated cardiomyopathy and elevated levels of catecholamines in the blood and urine. The presence of a retrovesical pelvic mass in contact with the right vaginal dome was determined by imaging studies. A diagnosis of malignant extra-adrenal pelvic paraganglioma with lymph node metastases was reached through biopsy and the surgical resection of subsequent local recurrences. Paragangliomas are usually located in the paravertebral zones from the base of the skull to the retroperitoneum and are benign in 90% of cases. This kind of neoplasia is uncommon in paediatrics, especially those located in the pelvis. In cases of masses of a gynaecological origin, a differential diagnosis should be considered, and a histological and immunohistochemical study is essential in certifying the diagnosis. Paraganglioma is a rare neuroendocrine neoplasia uncommon in the paediatric population originating in regional structures derived from neural crests. It is considered that between 10% and 20% of cases are diagnosed in the paediatric age group, with an estimated incidence of 0.011 for each 100,000 children under 18 years of age. Of these, approximately 12% are considered malignant . AccordiThe subject is a 13-year-old female patient with a history of arterial hypertension, tachycardia, and cardiomegaly. In the clinical evaluation a palpable lump was found in the hypogastrium. Full laboratory tests were requested and the following relevant data were found: adrenaline and noradrenaline elevated in blood and urine. An ultrasound was carried out revealing an ovoid, heterogeneous solid mass of 4.4 × 3 cm in contact with the vagina and below the uterus, causing an effect of mass on the bladder. In order to determine the nature, characteristics, location, and relationships of this mass, magnetic resonance imaging (MRI) was requested and this reported an image of 4 × 6 × 4 cm, located in the retrovesical and right vaginal parauterine region in contact with the posteroinferior wall of the bladder, which it deforms . It was Malignant extra-adrenal paraglangliomas are rare neoplasms, with less than 3% reported in the literature. Approximately 75% of the cases are sporadic and 25% are hereditary, associated with a family cancer syndrome such as multiple endocrine neoplasia type-2, Von Hippel–Landau syndrome and neurofibromatosis type 1. Paraganglioma is the term applied to tumours that originate in the autonomic nervous tissue regardless of location. They are usually located in the paravertebral area and with a thoracolumbar distribution, from the upper cervical region to the pelvis. Tumours of the head, neck, and anterior mediastinum are associated with the parasympathetic nervous system, whereas those of the posterior mediastinum, abdomen, and pelvis originate in the sympathetic nervous system , 9. DespThe majority of cases are diagnosed in the third to fifth decades of life, with paediatric cases being uncommon, and with those located in the pelvis being even less common. They usually present as pain and/or abdominal mass. Approximately 15% are asymptomatic and present as incidentalomas. They are frequently benign (90% of cases), while 10% are potentially malignant, as in the case presented. Clinical and imaging parameters are insufficient to predict the biological behaviour of these neoplasms and the presence of distant metastases is the only histopathological parameter of malignancy. The presence of a malignant paraganglioma in the paediatric age group makes it necessary to define the biochemical phenotype and family history, since it may be part of a syndromic condition, as in the case of mutations in the gene that codes for the enzyme succinate dehydrogenase B subunit SDHB), C (SDHC) or D (SDHD). It is considered that about 25% of the cases are carriers of mutations in the germinal line –13. PrimParagangliomas of the abdominopelvic cavity are neoplasias that are particularly uncommon in paediatric age groups. Clinical findings and imaging studies are variable and can even present as asymptomatic even when they reach a large size. It is important to note that in cases of pelvic locations in female patients a differential diagnosis should be carried out with masses of gynaecological origin, certifying the diagnosis with the surgical resection of the same and a subsequent histopathological and immunohistochemical examination.The authors declare that they do not have any conflicts of interestAll authors participated in the search for bibliography and of the preparation of the manuscript. All authors approved the final version thereof.


Muscle biopsy was also compatible with mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes and DNA analysis also concluded it. Since muscle biopsy is less invasive for patients compared to endomyocardial biopsy, cardiologists need to consider it. The diagnosis of mitochondrial cardiomyopathy is helpful because it is a genetic condition and also for consideration of device therapy, as well as management for acute crisis.We report a case with 46-year-old man diagnosed with mitochondrial cardiomyopathy in the dilated phase of hypertrophic cardiomyopathy. Since cardiac magnetic resonance imaging, beta-methyl-p- Mitochondrial disease is a heterogeneous group of multisystemic diseases due to mutations in nuclear or mitochondrial DNA. Although multimodalities aid the diagnosis, the diagnosis of mitochondrial cardiomyopathy is challenging because histological analysis is sometimes needed –3. We rehttps://doi.org/10.1155/2017/9473917). Serum lactate and pyruvate levels and cerebrospinal fluid lactate and pyruvate levels were elevated . Cardiac magnetic resonance imaging (MRI) showed no late gadolinium enhancement (LGE); positron emission tomography/computed tomography (PET-CT) showed no remarkable findings  myocardial scintigraphy revealed diffuse reduced uptake, except on the lateral wall  with an ejection fraction of 52%. His mother had died at the age of 50 due to dilated cardiomyopathy and had a history of diabetes mellitus. His brother also suffered hearing loss. The patient was a nonsmoker with no history of hypertension. His electrocardiogram revealed normal sinus rhythm and complete left bundle branch block (QRS duration: 128 ms) . B-type  Figures . Beta-meral wall . Coronarral wall . Electroral wall . Since tral wall . FinallyThe diagnosis was concluded as mitochondrial cardiomyopathy and MELAS. After treatment with intravenous dobutamine for reduced output, he was given angiotensin-converting enzyme inhibitor, beta-blocker, spironolactone, coenzyme Q10, and carnitine. Since he complained of nausea after dobutamine therapy, we performed a gastroscopy and stomach biopsy. Electron microscopic examination showed no mitochondrial deformation . At 7 mo123I-BMIPP myocardial scintigraphy revealed diffuse reduced uptake except in the lateral wall and fatty acid metabolism disturbances, but it was indefinite because another study insisted 123I-BMIPP hyperaccumulation is related to mitochondrial cardiomyopathy [We report a case, diagnosed with mitochondrial cardiomyopathy in the dilated phase of hypertrophic cardiomyopathy. Although 30% of cases of mitochondrial myopathy showed LGE, the MRI in our case revealed no significant findings in contrast to a previous study . PET-CT myopathy . Muscle myopathy , cardiolmyopathy . In our myopathy –8.We report a case of mitochondrial cardiomyopathy presenting as dilated phase of hypertrophic cardiomyopathy diagnosed with muscle and endomyocardial biopsy and genetic analyses. Since muscle biopsy is less invasive for patients compared to endomyocardial biopsy, cardiologists need to consider it regardless of muscle weakness.Online Video 1: Transthoracic echocardiography, long axis view.Online Video 2: Transthoracic echocardiography, short axis view. Online Video 3: Transthoracic echocardiography, 4 chambers view.


Dear Editor,We report a case of a 41-year-old woman with lower abdominal pain and vaginal bleedingwith negative ultrasound scan except for fibroids, and elevation of serumβ-hCG . Differential diagnoses included ectopic pregnancy,early pregnancy failure, very early ongoing pregnancy and molar pregnancy.MRI showed an enlarged uterus with central heterogeneous T2 hyperintensity distending theendometrial canal, demonstrating reticular enhancement, concerning for gestationaltrophoblastic disease. There was also distortion of the junctional zone with broadintermediate-to-low signal on T2-weighted images. The variable appearance of myometrialthickness especially on postcontrast images, showing a homogeneous myometrium,facilitated the diagnosis of contractions . The pat,2. HM is the mostcommon manifestation of GTD (85%) and by definition noninvasive and confined to theendometrium. A HM that invades the myometrium is termed invasive mole, and is composedof HM villi within the myometrium. Chorioadenoma destruens is a locally invasive(myometrium) manifestation of complete HM that represents 13% of cases of GTD. Twopercent of complete HM cases are described as choriocarcinoma, which is locally invasiveand potentially metastasizing. These three entities produce peculiarly high levels ofβ-hCG, while placental site trophoblastic tumor causes a rise in humanplacental lactogen levels, and less elevated β-hCG levels,4. Clinical assessment is difficult early in the course of the disease,as few clinical characteristics are present to distinguish it from a normalpregnancy.Gestational trophoblastic disease (GTD) arise from placental trophoblastic tissue afterabnormal fertilization and comprises a spectrum of disorders from the pre-malignantconditions of partial HM and complete HM to the malignant invasive mole, choriocarcinomaand the very rare placental site trophoblastic tumor ,5. Early manifestations appear as a softtissue cystic mass with high T2 signal intensity. In the second trimester these lesions tend to distend theendometrium giving a "cluster of grapes appearance". Typically HMs are similar orslightly higher in T1 signal intensity than the adjacent myometrium. Contrast-enhancedMRI show areas of focal enhancement that relate to the amount of active trophoblastictissue and also to β-hCG levels. Marked early enhancement indicates active disease in the form ofviable trophoblastic tissue.Pelvic MRI is often used as a problem-solving tool in equivocal or complicated cases ofGTD, especially in the first trimester, or to assess the degree of myometrial invasionand surrounding tissues. Myometrial contractions are seen as a bulge of the myometrial wallusually along with a region of low T2 signal intensity in the myometrium. They aretransient and tend to disappear on subsequent data acquisitions, as observed in our case. In thesetting of endometrial tumor, radiologists should be aware of this phenomenon to avoidover-diagnosis and over-staging by misdiagnosing uterine contraction with myometrialextension or invasion.In the setting of GTD, identification of myometrial invasion is crucial for diagnosis andstaging. Uterine tumors associated with high serum β-hCG have a highincidence of myometrial contractions


Congenital uterine anomalies like bicornis or bicornuate uterus are relatively rare in sub-Saharan Africa. They are associated with an increased rate of spontaneous abortion, preterm delivery, and infertility. The occurrence of bicornis bicollis uterus with unilateral cervical atresia is exceptional and its management is controversial. We hereby report a rare cause of chronic pelvic pain in a Cameroonian teenager due to unilateral obstructive hematometra and hematosalpinx in the non-communicating horn of a bicornis bicollis uterus.A 13-year-old premenarchal non-virgin female presented with chronic and severe cyclical crampy pelvic pain. On clinical examination, she had a perforated hymen, a single vagina, and one uterine cervix. A two-dimensional pelvic ultrasonography revealed hematometra but missed out the underlying anomaly. Failure to drain the hematometra by serial cervical dilatations prompted an exploratory laparotomy which revealed: bicornis bicollis uterus with a right rudimentary uterine horn communicating with the vagina and a left non-communicating uterine horn distended by hematometra due to a homolateral cervical atresia. She underwent utero-vaginal canalization and a left hemi-hysterotomy with drainage of the hematometra. The postoperative period was uneventful. Regular cyclic menses occurred thereafter beginning at the first postoperative month. She had complete resolution of symptoms without recurrence after six months.Due to the risk of compromised fertility from bicornis uterus and the diagnostic challenges akin to resource-limited settings, we highlight the need for a high index of suspicion by healthcare providers when faced with chronic pelvic pain in premenarchal adolescents. Bicornuate uterus (BU) is a congenital uterine anomaly with a prevalence of 0.4% in the general population . It is oContrary to high-income countries where epidemiological studies have been carried out on these anomalies , the rel0P0 presented with severe cyclical pelvic pain of gradual onset for a duration of 15 months. The pain was constricting in nature, predominant in the left iliac fossa, radiating to the back and perianal region and unremitting to analgesics and anti-spasmolytics. There was no fever, no urinary or gastrointestinal symptom. She had interrupted her education eight months prior to consultation due to the incapacitating nature of the pain.A 13-year-old female student GShe was premenarchal, had pubarche and thelarche at the ages of 9 and 11 years respectively. She had no chronic illness and had never undergone surgery. Her psychosocial and family histories were unremarkable. She had first coitus at the age of 11 years.On examination, she was ill looking and had Tanner stage IV secondary sexual characteristics. Her temperature was 38.1 °C (100.58 °F) and other vital signs were within normal limits. Her conjunctivae were not pale. Abdominal examination revealed tenderness at the supra-pubic region and left iliac fossa without guarding. A moderately tender, rubbery, roughly spherical, supra-pubic pelvic mass of about 7 cm (longest diameter) was palpable. The physical appearance of her external genitalia was normal and she had a perforated hymen. On speculum examination, the vagina and cervix were normal. There was mild cervical motion tenderness, the pouch of Douglas was empty and the uterus was of normal size. Supra-pubic tenderness hindered clinical evaluation of the adnexae. Digital rectal examination and other systemic examinations were non-contributory. A working diagnosis of cervical stenosis complicated by cryptomenorrhoea was made and as differential diagnoses: chronic pelvic inflammatory disease and a pathologic ovarian cyst were advocated.A two-dimensional pelvic ultrasound scan demonstrated the presence of hydrometra of 225 ml Fig.  and bila. The association of BU with cervical atresia is unusual because BU is often a non-obstructive type of Mullerian anomaly. Only sporadic cases similar to this one have been reported along with their surgical management [Congenital uterine anomalies are incidental findings. A recent systematic review showed a prevalence of 5.5% in the general population, with a prevalence rate of 0.2%- 0.6% accounted by BU . BU arisnagement . The occnagement , 8. Pelvnagement . Alternanagement  or symptnagement . The resnagement , 13. Ournagement . Howevernagement . Our pat. Historically, due to the high postoperative mortality rate and complications of utero-vaginal canalization, namely re-stenosis of the neo-ostium, low postoperative fertility rate and severe infection by ascending route, some authors advocate for hysterectomy even in young patients [The treatment of BU with cervical atresia is controversial, consisting of surgical techniques like hysterectomy or utero-vaginal canalization , 4. The patients , 16. Howpatients , 17. Sompatients , 18. TogFinally, fertility prognosis is debatable as successful term pregnancies by natural or artificial insemination have been reported after surgical correction , 10, 19.We have reported the first case of concomitant bicornis bicollis uterus with unilateral cervical atresia in Cameroon. Although limited resources precluded proper investigation with magnetic resonance imaging, we have shown that utero-vaginal canalization could yield favourable postoperative outcomes. This would need to be further explored in large multi-centre clinical trials in our setting. Health care personnel should have a high index of clinical suspicion for this congenital uterine anomaly as a potential differential diagnosis in premenarchal Cameroonian adolescents with unremitting cyclical chronic pelvic pain. Robust and advanced imaging modalities when available should help confirm clinical suspicion. The benefits of a timely diagnosis and treatment of such congenital uterine anomalies cannot be overemphasized in a setting already burdened with elevated female infertility.


An osteoblastoma is an uncommon benign but painful tumor, typically found on the axial skeleton or on long bones in the case of young patients. Some cases of humeral osteoblastomas have been described in literature but not in men older than 30. We report the case of a painless bone tumor on the humerus of a 52-year-old patient. The CT scan shows a 30 mm hypodense lacunar formation, surrounded by thickened cortical bone resembling an osteoid osteoma. The anatomopathological and immunohistological analyses support the thesis of an osteoblastoma. A course of radiological monitoring without surgical resection was adopted. This unusual case introduces the possibility of carrying out a differential diagnosis with an osteosarcoma and raises the question of the treatment that should be adopted. An osteoblastoma is an uncommon benign but painful tumor usually found on the axial skeleton or on long bones in the case of young patients. The presence of a painless osteoblastoma on the humerus is unusual in a patient over the age of 50.A 52-year-old male consulted for sudden swelling in the left deltoid region without his being aware of any recent traumatism. The clinical examination confirmed the painless swelling, which did not cause functional disability, suggesting a rotator cuff tear. An ultrasound did not show anything in particular. An X-ray found a hypodense lesion in the cortical bone on the upper third of the humerus . The MRIA CT-guided biopsy was carried out which identified a fibro-osseous lesion with osteoid bone surrounded by osteoclasts in the pathological anatomy. Between the osteoid bones, fairly dense fibroblastic tissue made up of spindle-shaped cells, without cytonuclear atypia, was identified. A large number of foamy macrophages and some giant multinucleate cells were also observed. There was an abundant blood supply. The immunohistochemical staining was positive with antigalectine 1, around the macrophagic cells with anti-CD68, and negative with anti-CDK4 and -MDM2. This was suggestive of an osteoblastoma without cytological evidence of malignancy.The patient underwent surgery on the rotator cuffs and was then placed on a course of clinical and radiological surveillance every 6 months. There was nothing of note in the clinical check-up 6 months later and the patient did not complain of any residual pain. The scanner showed an indented humeral head with incisions in the front cortical bone at two points, without a “nidus”. At the 1-year check-up the MRI scan found a slightly raised area resembling a post-operative reformation. The decision not to operate on the osteoblastoma was maintained with image monitoring every 6 months, except in the case of an adverse clinical development.Five years after this episode, the patient is still under surveillance. He does not present any functional problems or radiological changes of the existing lesion.Our patient raises two issues: the rupture of the rotator cuff and the bone lesion.The rupture of the rotator cuff does not cause any functional disability in our patient. The absence of correlation between the anatomical lesion and the significance of the symptoms is found in literature . In our Osteoblastomas are an uncommon type of benign osteoid osteoma-like tumor and represent 1% of all bone tumors . They deIn our case, a 52-year-old man with a painless bone lesion, unusually on the humerus, raises the question of the diagnosis and that of the development of the lesion. Is this an osteoid osteoma or an osteoblastoma? Is there a risk that it will develop into a malignant tumor?In the first instance, the diagnosis is based on the X-ray. In long bones, osteoblastomas are spindle-shaped osteolytic lesions that can break through the cortical bone with a moderate peripheral halo. Calcification can be found at the center of the osteolysis giving it a characteristic appearance . In our The positive diagnosis was obtained through a bone biopsy carried out under scan control. The anatomopathological analysis provided arguments in favor of an osteoblastoma due to the abundance of osteoblasts. With regard to osteoblastomas, current cytogenetic research has found specific markers that do not affect the form of treatment suggested and the prognosis .Surgical treatment is necessary in the event of disabling pain or the aggressive growth of the tumor. It can be carried out by means of curettage or complete resection, with or without bone implants. Surgical resection seems to be more effective in avoiding relapse but certain studies suggest a risk of malignant transformation after curettage . TreatmeOsteoblastomas are benign tumors that in rare cases can develop into osteosarcoma- or osteochondroma-type tumors , 14. ThiWe have presented a rare case found in the course of a general medicine consultation. In cases of bone lesion or in age groups at risk, complementary tests are necessary. A biopsy is needed in order to obtain a definitive diagnosis. Even if clinical and X-ray tests suggest that the lesion is benign, we cannot always make the distinction between an osteoid osteoma and an osteoblastoma. This diagnosis is important in order to determine the course that should be followed. The risk of malignant transformation justifies active monitoring.