Pancreatoblastoma is a rare paediatric malignant neoplasm. The treatment of choice is complete surgical resection. However, it is often unresectable due to its large size, local infiltration or distant metastasis. Since the condition is rare, there is currently no standard treatment regimen. We outline the case of a 4-year-old child who presented with abdominal pain and distention, together with an enlarged liver and elevated serum α-fetoprotein levels. Imaging studies showed the presence of an abnormal pancreatic tumour and multiple nodular lesions in the liver, the biopsies from which led to a diagnosis of pancreatoblastoma. In this case, the patient received cycles of neoadjuvant chemotherapy, combining cisplatin and doxorubicin. The patient subsequently underwent scheduled surgery in which the primary pancreatic lesion was resected, obtaining a circumscribed and nodular specimen measuring 7 × 6 cm and weighing 150 g. Given the extent of the metastasis, the child is currently awaiting a liver transplant. Pancreatoblastoma is the term given to rare malignant neoplasms  that mimA 4-year-old male Caucasian patient with no prior family history of neoplasms presented in fair general condition with weight loss and loss of appetite, which, according to his parents, had developed over 2 months. Clinical evaluation showed evidence of abdominal pain and distention in addition to hepatomegaly. A full work-up was requested, significant findings were anaemia, very high levels of α-fetoprotein , low levels of chorionic gonadotropin subunit beta (1.9 mIU/mL) and elevated levels of lactate dehydrogenase . Tests on the levels of catecholamines in the blood and urine were negative. An abdominal ultrasound was performed, which showed irregular hepatomegaly with a heterogeneous echotexture due to the presence of multiple, randomly distributed echogenic nodules, suggestive of metastasis. The pancreas could not be evaluated. This was followed by an abdominal and pelvic CT scan with and without contrast dye: this showed a heterogeneous tumour in the body and tail of the pancreas that had defined borders, hypodense areas inside (suggestive of necrosis) and an apparent pseudocapsule. This measured: anteroposterior diameter 75 × transverse diameter 57 × longitude 101 mm, displacing the left kidney and the splenic artery in the head and rear directions, the left ureter in the dorsal direction and the bowel in the caudal direction. Enlarged heterogeneous liver, due to the presence of multiple hypodense nodules. The full body bone scan pathology did not show focal uptake. The adrenal glands showed no significant changes.The patient’s condition deteriorated: he presented an acute abdomen requiring surgery, for which an exploratory laparotomy was performed. This revealed a hepatic nodular lesion in the left lobe, with surface erosion and bleeding, from which biopsies were taken. The histopathological study with Haematoxylin–Eosin (H & E) staining showed limited liver trabeculae, infiltrated by a poorly differentiated malignant neoplastic proliferation consisting of medium-sized round or polygonal cells with large cytoplasma, with areas of necrosis and haemorrhage. Immunohistochemistry procedures were run, revealing neoplastic cells: positive for vimentin, Cytokeratin (AE1–AE3), β-catenin, Ki-67 (in 80% of the neoplastic nuclei) and, focally, carcinoembryonic antigen. In contrast, the tests were negative for neuron-specific enolase, Chromogranin and Hep Par-1. The earlier findings, together with the patient’s clinical context, suggested liver metastasis due to pancreatoblastoma.2 and doxorubicin 21 mg/m2.The patient went into intensive care and, upon returning to the ward, started a course of chemotherapy, using cisplatin 56 mg/mFollowing neoadjuvant treatment, the patient underwent schedule surgery for excision of a pancreatic tumour. The surgical approach was through the lesser sac, locating it in the tail and body of the pancreas. We proceeded to the lower margin, releasing the relevant vessels, to the posterior for unaffected pancreatic tissue and, finally, to the upper margin respecting the splenic vessels. A circumscribed, nodular lesion of 7 × 6 cm and weighing 150 g was obtained in the aforementioned procedure (pancreatectomy of the body and tail). The site of incision had a yellowish surface with solid areas and a friable central section. Histological sections showed the presence of a pancreatoblastoma, with large areas of necrosis and two residual peripheral nodules. Additional immunohistochemical techniques demonstrated the membrane positivity for E-cadherin and vascular endothelial growth factor (VEGF).At present, the patient is receiving medical follow-up and is in a liver transplant programme.et al [et al [Frable et al  were thel [et al  also madl [et al , 7, whicl [et al .Pancreatoblastoma accounts for 0.2% of all neoplasms in the pancreas, being most frequent among children (mean age of 5 years) , 10. In Macroscopically, the lesion is usually enclosed, lobed and solid with a heterogeneous, necrotic, haemorrhagic cut surface and a mean diameter that can reach up to 11 cm , 17. MicImmunohistochemical techniques show diffuse expression of Cytokeratin membrane (AE1–AE3) and epithelial membrane antigen, while in areas of acinar differentiation one tends to find typical pancreatic exocrine enzymes such as trypsin, chymotrypsin and lipase, the latter to a lesser extent . In addiet al [In the study conducted by Bien et al , completet al , 12, 20.et al .et al [Finally, the work done by Dhebri et al  outlinesPancreatoblastoma is a rare malignant neoplasm, which usually presents as a slow-growing abdominal mass and with high serum levels of α-fetoprotein. This should be always considered in a young child. It represents a real diagnostic challenge for oncologists, surgeons and pathologists. For this reason, it is necessary to gather as much clinical and additional information as possible when a surgical specimen is taken. The treatment of choice is a complete resection of the primary neoplasia, although this can also include neoadjuvant chemotherapy and, in cases with massive metastasis, liver transplantation. The case study illustrates the characteristics of the disease and the strategies currently available to approach a patient’s treatment.The authors have no conflicts of interests to declare.All authors participated in the bibliographical research and preparation of the manuscript. All authors approved the final version of the same.The authors declare that the work submitted has not received specific financial support from the public sector, commercial bodies or non-profit entities.


Schwannomas are rare tumors, usually benign, originating from the Schwann sheath of the peripheral or cranial nerves. They are an extremely uncommon cause of adrenal incidentaloma. It is difficult to diagnose adrenal schwannoma preoperatively by imaging alone due of their aspect suspect on it. We report a case of adrenal mass discovered incidentally in a 41 years old patient who presented an atypical abdominal pain. Biological assessment was unremarkable, and imaging studies did not show strict imaging criteria for a typical adenoma. A surgical excision of the adrenal incidentaloma was decided. Histopathological examination and immunohistochemical staining of the excised specimen revealed a benign schwannoma. Schwannomas are unfrequent tumors, usually benign, originating from the Schwann sheath of the peripheral or cranial nerves . VisceraA 41 years old patient, presented with epigastric pain and indigestion since 1 year. Physical examination and ultrasound abdomen were normal. A symptomatic treatment was prescribed without improvement. On physical examination the abdomen was soft, blood pressure was 130/85 mm Hg and pulse rate was 72 beats/min; sinus rhythm was normal. Blood count and biochemistry analysis were within normal limits. Computed tomography of the abdomen showed a left adrenal mass (2.5 X 3.4 cm); with a spontaneous density of 20 Hounsfield units (UH). The relative and absolute washouts were 10 % and 16 % respectively . Based oThe greater number of the tumors originating in the adrenal medulla is pheochromocytoma, neuroblastoma, or ganglioneuroma. Adrenal schwannoma is rarely encountered, even though it similarly originates from the cells of the neural crest. Its first description was done in 1908 by Verocay as neurinoma . In 1932The present case suggests that a preoperative diagnosis of adrenal schwannoma is difficult. Surgical excision can provide the benefit of a definitive diagnosis which can only be made by histological and immunohistochemical evaluations.The authors declare no competing interests.


The lung is the organ most commonly affected by primary synovial sarcoma. Intratumoral calcification is less common in this organ versus soft tissue. Meanwhile, the presence of calcification in a lung nodule reduces the risk of lung cancer. Here, we report a case of pulmonary synovial sarcoma which manifested as a nodule with calcification, depicted on computed tomography (CT). A 52‐year‐old asymptomatic male was referred to Saitama Medical University International Medical Center and CT revealed a well‐defined nodule (1.8 cm), with punctate and eccentric calcification in the right lower lobe. Enhanced CT and 18F‐fluorodeoxyglucose positron‐emission tomography suggested a malignant tumor, and surgery was performed. Histology provided a preliminary diagnosis of monophasic spindle‐cell synovial sarcoma with hyalinized collagen bands and calcifications. Genetically, the presence of the SYT‐SSX2 fusion gene was consistent with the features of this disease. We conclude that primary pulmonary synovial sarcoma should be listed as a differential diagnosis for solitary pulmonary nodules with calcification. Significant findings of the study: Primary pulmonary synovial sarcoma can manifest as a nodule with calcification shown on computed tomography, and gene analysis is useful in reaching a definitive diagnosis for this disease.What this study adds: Primary pulmonary synovial sarcoma should be listed as a differential diagnosis for solitary pulmonary nodules with punctuate and eccentric calcification.Synovial sarcomas predominantly arise in the deep soft tissue of the lower and upper extremities; however, they appear in almost all anatomic locations. The most commonly involved visceral organ is the lung. The name synovial sarcoma is described as a misnomer, owing to the lack of evidence regarding synovial differentiation.During a follow‐up examination of post‐traumatic thoracic aortic aneurysm treated by a stent‐graft 17 years earlier, a 52‐year‐old asymptomatic male was referred to Saitama Medical University International Medical Center. The patient was undergoing treatment for hypertension, hyperlipidemia, and diabetes mellitus. Noncontrast chest CT incidentally revealed the presence of a well‐defined nodule measuring 1.8 cm, eccentrically with focal calcification in the right lower lobe  Primary pulmonary synovial sarcoma can manifest as a nodule with calcification shown on CT; and (ii) gene analysis is useful in reaching a definitive diagnosis for this disease. In addition, we focused on radiological and histological calcification findings for primary pulmonary synovial sarcoma.Synovial sarcoma is derived from multipotent stem cells capable of differentiating into mesenchymal and/or epithelial cells. Histologically, synovial sarcoma can be classified into the biphasic, monophasic fibrous or spindle‐cell type, and poorly differentiated type.The first important clinical finding is that pulmonary synovial sarcoma can manifest as a nodule with calcification shown on CT. Focal calcifications – rarely accompanied by ossification – shown on radiographs or CT scans are identified in ≤30% of cases with soft tissue synovial sarcoma.Regarding the imaging of lung nodules, from the CT perspective, intranodal calcifications are more commonly observed in benign lesions: inflammatory granulomas caused by mycobacterial or mycotic infections, or a benign tumor . In contrast, calcifications are less frequently detected in primary lung cancers (6.0%).SS18 gene on chromosome 18 and one of the SSX genes  on the X chromosome are fused, leading to the generation of the SS18‐SSX1, SS18‐SSX2, or – rarely – SS18‐SSX4 fusion oncogenes.SS18 split‐signal in almost all tumor cells. The subsequent RT‐PCT analysis demonstrated that the present case harbored the SS18‐SSX2 fusion oncogene. Regarding the association of gene fusion and histology, almost all biphasic and monophasic types have been shown to harbor the SS18‐SSX1 and SS18‐SSX2 fusion oncogenes, respectively.The second important clinical finding is that gene analysis was useful in the diagnosis of this disease. In the present case, we considered the presence of a synovial sarcoma based on morphological and immunohistochemical findings. However, it was necessary to differentiate this type of sarcoma from others which are categorized as malignant spindle‐cell tumors . Synovial sarcoma is characterized by the t   chromosome translocation, which is exclusively observed in this tumor. The Primary pulmonary synovial sarcomas are rare, comprising <0.5% of all primary lung malignancies.In conclusion, primary pulmonary synovial sarcoma can manifest as a nodule with calcification shown on CT. In addition, gene analysis is useful in reaching a definitive diagnosis for this disease. Primary pulmonary synovial sarcoma should be listed as a differential diagnosis for solitary pulmonary nodules with punctuate and eccentric calcification. When considering synovial sarcoma based on CT findings and immunohistochemical analysis, oncology surgeons should complement the examination by performing fluorescence in situ hybridization analysis. Further investigations are warranted to determine whether a primary pulmonary synovial sarcoma with calcification may be more frequently detected.The authors report no conflict of interest.


Primary lymphoma that arises from the intestine is an uncommon malignant tumour, while intestinal fistula caused by primary lymphoma is even rarer. Non-specific clinical performance makes early diagnosis difficult, although imaging modalities might play an essential role in the detection of intestinal fistula.Patient 1: A 60-year-old male hospitalized with diarrhoea and abdominal pain for seven months underwent computed tomography enterography (CTE) that demonstrated ileum internal fistula and ileac-sigmoid colon fistula. Ultrasound (US) showed small intestinal wall thickened and development of a fistula of the sigmoid colon due to malignance. Patient 2: A 43-year-old male presented with abdominal pain and diarrhoea lasting one year. US revealed a fistula between the sigmoid colon and the ileum, and CTE showed that the wall of the partial sigmoid colon was abnormally thickened and enhanced with an ileal-sigmoid fistula that strongly suggested the diagnosis of lymphoma.Both the two patients were diagnosed as intestinal fistula caused by primary non-Hodgkin's intestinal lymphoma.The patient 1 underwent surgery followed by chemotherapy. The patient 2 accepted chemotherapy.Two patients’ general conditions remained stable and the imaging revealed no recurrence after follow-up of about 12 months.Cross-sectional imaging, such as US and CT, plays an essential role in intestinal lymphoma fistula diagnosis. A tumor bowel fistula is an uncommon but dangerous complication in the advanced stages of primary intestinal lymphoma. Because the clinical symptoms and signs are not specific, imaging modalities can help in detecting this complication. Few intestinal fistulas caused by primary lymphoma have been reported.,4 In this report, we describe the clinical manifestation, imaging features, treatment, and prognosis of 2 cases of intestinal fistula that were complications of small intestine lymphoma.Primary lymphoma is a rare malignant tumor in the gastrointestinal (GI) tract that accounts for approximately 1% to 4% of all GI malignancies.22.12, and an approximately 3-cm-diameter, relatively hard, slightly movable mass was palpable in the left lower abdomen without obvious tenderness or superficial lymphadenopathy. Laboratory examination showed the following positive findings: C-reactive protein level (CRP) was 12.14 mg/L↑  and fecal occult blood (OB) was positive (+). The blood routine, erythrocyte sedimentation rate (ESR), set of tumor markers, antinuclear antibody spectrum (ANAs), and inflammatory bowel disease antibody spectrum showed no abnormalities. Computed tomography enterography (CTE) demonstrated that the regional 6th small intestine wall was enhanced with multiple air pockets inside the involved bowel. The lesion abutting the ileocecal junction and sigmoid colon had a distorted contour  was 20.1 kg/m2.22, and no mass could be distinctly palpated on his scaphoid abdomen. Laboratory examinations showed the following blood and biochemical findings: 95 g/L hemoglobin, OB (+) stool, and 20.24 mg/L CRP, negative for the entire set of tumor markers and negative for T-SPOT.TB (tuberculosis). The transabdominal US demonstrated that the intestinal wall of the sigmoid colon was irregularly thickened and had a loss of normal construction, presenting a hypoechoic mass as the rough serosa. Increased blood flow signal was also detected in the intestinal wall. The sigmoid colon was found adhered to the abutting pelvic small intestine. A fistulous communication was confirmed when intestinal content was moving between the sigmoid colon and the ileum during a real-time dynamic US scan , followed by the large bowel (22%) and the gastric tract (16%). According to the literature, intestinal fistula caused by lymphoma could affect the adjacent loops, the bladder and even the aorta.–9Spontaneous tumor bowel fistulas are generally reported to be caused by cervical, ovarian, and colon cancers. During this period, the patients are unlikely to present symptoms that are typical of fistula formation and, thus, are not diagnosed. The most common presenting symptoms of the primary small intestinal lymphoma are abdominal pain (84%), weight loss (81%), and diarrhea (39%), which are symptoms that also resemble other GI diseases. Only 23% of patients present with features of small intestinal perforation. Similarly, in the 2 cases mentioned above, abdominal pain and diarrhea were the 2 primary symptoms of the patients. The possible reasons why their perforation failed to be recognized are the poor general condition or that the lesion was confined. However, factors that may suggest the possibility of perforation include sudden increased pain or hematochezia. Patients with intestinal internal fistula will present with obstinate abdominal pain, intractable diarrhea, and malnutrition. In our case, the persistent diarrhea might be a result of colonization of the small intestine with colonic flora from the sigmoid colon. It is possible that the fistula between the small and large intestine offers a shortcut, causing intestinal content to fail to be well digested and absorbed, similar to short bowel syndrome.Fistula-formation is a chronic and long-term process. In the early period of primary intestinal lymphoma, the tumors are confined in the submucosa; with the progression of the course of lymphoma, tumors gradually invade the serosa and perforate into the mesentery, finally resulting in a confined abscess or an intestinal fistula after the lesions subsequently infiltrate adjacent intestinal walls. Certain imaging findings, such as a bulky mass with small intestinal loops wrapped around it, an air-contrast level and a fistulous tract with the tumor, may strongly suggest the formation of an intestinal fistula. A CT scan can delineate abnormally enhanced thickened intestinal wall and detect the site, shape and tract of the fistula. Because of its advantage allowing multiple sections to be observed, transabdominal US is more sensitive than CT for demonstrating the tract where the fistula is encountered; not only does US show the hypoechoic, thickened and rich-blood-flow intestinal wall, but it also enables the observation of dynamic movements of stiff and narrow lumen. US, CT, and magnetic resonance imaging (MRI) have high sensitivity and specificity for the diagnosis of intraabdominal fistulas, with similar diagnostic accuracies. Concerning the tumor bowel, differential diagnoses of a gas-forming infection or tumor necrosis are important if the imaging tests reveal the presence of gas alone in the tumor. A tumor-bowel fistula could be diagnosed if the tract between the involved bowels is detected or if there is air inside the lesions.CT and MRI are able to provide valuable information for staging the malignancy as well as assisting in preoperative planning.; it is important in assessing disease activities, including both inflammation and necrosis. We can take full advantage of PET/CT to evaluate the prognosis and curative effect of patients after chemotherapy. For such patients, as observed in case 1, aiming at the targeted involvement site, the combination of PET/CT before chemotherapy and contrast-enhanced CT plays a vital role in the estimation of surgical indication and the optimal timing of surgery. However, the efficacy of PET/CT for evaluating the intestinal fistulization caused by lymphoma partially depends on the CT protocol. As in our 2 cases, PET/CT could not directly detect the fistula without the use of bowel contrast in the protocol.PET/CT is widely applicable for both the localization and viability of malignancies However, compared to other noninvasive imaging examinations, the enteroscope sometimes fails to reach the fistula because of the narrow and inflamed intestinal lumen. Biopsy under enteroscopy often yields a negative result because of deep lymphomatous lesions that increase the risks of bleeding and perforation. As in the first case presented here, intestinal lymphomas often do not receive a definitive diagnosis until the pathological results are obtained after surgery.Enteroscopy permits direct visualization of the mass and contrast of the mucosa of both intestinal segments. Chemotherapy is a good option for patients with advanced intestinal lymphoma. Since data about incidents of postoperative complications are limited, the true value of selective operation for prognosis cannot be assessed. The 2 patients in this report tolerated the undesirable effects of chemotherapy well, and their conditions significantly improved in the follow-up period.The prognosis for such cases is very poor; the survival rates associated with perforation at 6 months and 1 year was reported as 27.5% and 0%, respectively. Poor prognosis is attributing to several factors, which including the fast-growing T-cell lymphoma and resistance to chemotherapy regimens.4Intestinal fistula is a relatively rare complication of primary intestinal lymphoma, especially in patients who do not receive chemotherapy. Cross-sectional imaging modalities, such as US and CT, play an essential role in intestinal lymphoma fistula diagnosis as well as in treatment follow up.Methodology: Miaoqian Wang, Qian Yang, Wei Liu.Resources: Wenbo Li, Hong Yang, Weixun Zhou.Software: Ji Li.Writing – original draft: Nan Zhuang, Qingli Zhu.Writing – review & editing: Qingli Zhu.


ABCs are expansile osteolytic lesions typically containing blood-filled spaces separated by fibrous septae. Standard treatment includes surgical resection or curettage and packing; however, for some spinal lesions, the standard approach is not optimal. One therapeutic strategy is to treat spinal ABC with an agent that targets a pathway that is dysregulated in a disease with similar pathophysiology. Denosumab, a human monoclonal antibody to RANKL is effective in the treatment of GCT's. Spinal ABCs are a therapeutic challenge and local recurrence is a concern. We report a case of aggressive recurrent ABC of dorsal spine in a 14-year old female with progressive neurologic deficit who underwent surgical excision and decompression with a recurrence in a short period for which a decompression and fixation was done. She had a recurrence after an asymptomatic period of 6 months and neurologic worsening. Having ruled out use of embolization and radiotherapy, a remission was achieved by treatment with Denosumab using the regimen for GCTs for a duration of 6 months. Follow-up MRI and CT scans at 24 months following inception of Denosumab depicted complete resolution and no recurrence. We conclude that Denosumab can result in symptomatic and radiological improvement in the recurrent locally aggressive ABC and may be useful in selected cases. Long-term results are mandatory to confirm the efficacy of Denosumab and to evaluate local recurrence after stopping Denosumab. ABCs are expansile lesions accounting for approximately 15% of all primary spinal tumours . MultiplGiant-cells that occur in ABCs are positive for markers of true osteoclasts ,7. The oA 14-year-old female presented with unsteady gait and back-pain. She had undergone two previous spinal surgeries in the recent past at another hospital. The previous medical-records showed that the girl developed insidious-onset, progressive spastic sensory-motor paraparesis. MRI of the dorsal-spine  suggesteTreatment was begun using the regimen described for treating GCT –11 120 . DenosumABCs are generally rare tumorous lesions of the spine, but can lead to devastating symptoms as they are capable of destructive growth. Common treatment options for ABCs include surgical resection, curettage and filling, selective arterial embolisation, total arterial embolisation or radiotherapy. For spinal ABCs, however, some of these options entail considerable risks. Sclerosant therapy was not considered in our patient as the cortices were eroded following previous surgical excisions, and hence there was a risk of seepage of sclerosant into neuro-vascular elements leading to catastrophic complications .Denosumab, a human monoclonal-antibody, binds to a cytokine receptor activator of nuclear factor-kappa Bligand (RANKL) and prevents the action of agonists acting through RANKL-receptors. This prevents the subsequent activation and proliferation of the osteoclasts . Seven pDenosumab has beneficial effects as in case of recurrent ABC described in this report. Surgical endeavours had failed twice and other proven modalities were ruled out. Previous failed surgeries had left the surgeons as well as the patient and relatives in despair with low hopes of further surgeries and in the outlook for newer horizons. We were unable to find a similar report of an aggressive refractory ABC of the dorsal spine in which deployment of Denosumab was successful.We conclude that the usage of Denosumab, although based on anecdotal experiences, may be an excellent option in cases not amenable to surgical interventions and frequent recurrences. Long-term results should confirm these preliminary outcomes to standardise the treatment-guidelines.


The chondroid syringoma is an extremely rare skin tumor most commonly found in the area of the head and neck region. Its rarity, potential for malignancy, and frequent misdiagnosis for other more common tumors can impart unique challenges in diagnosis and management. Diagnosis is usually revealed by excision followed by histologic examination. We report a case of a 42-year-old male with no prior medical history diagnosed with chondroid syringoma of the forehead and review the relevant literature. The chondroid syringoma is a rare benign skin appendageal neoplasm first described by Hirsch and Helwig in 1961 . ClinicaA 42-year-old man with no medical history presented with a firm, painless, mobile 2 cm nodule Figure . He repoChondroid syringoma, or a cutaneous mixed tumor, is an adnexal neoplasm of either apocrine or eccrine origin. It presents as a solitary, firm, dermal or subcutaneous nodule, usually between 0.05-3 cm . The lacIf there is a nodule in the head and neck region, although rare, the chondroid syringoma should be considered in the differential diagnosis. With the low incidence of these cutaneous tumors along with non-prominent clinical presentation, excision of the mass followed by histopathological examination is recommended for diagnosis and treatment.


Benign breast papilloma is currently managed with conservative management with close observation. In contrast, papilloma with high-risk or malignant lesions warrants surgical excision. The purpose of our study was to investigate magnetic resonance imaging (MRI) features of breast papilloma and to identify imaging diagnostic indicators for papilloma with high-risk or malignant lesions.MRI features of 175 surgically confirmed papillomas on 158 patients were retrospectively reviewed. The 175 cases included 132 cases of benign papilloma and 43 cases of papilloma with high-risk or malignant lesions. The MRI features of these lesions were classified into three types: mass, non-mass enhancement (NME), and occult lesion. The occult lesion was defined as the presence of only ductal dilation without any enhanced lesions on MRI. For a mass lesion, the mixed mass-NME lesion was considered if linear, segmental or regional enhanced lesion was found adjacent to the mass. Clinical and MRI features were compared by univariate and multivariate analysis between the benign papilloma and the papilloma with high-risk or malignant lesions.n = 135, 77.1%), and fewer as NME  and occult lesion . For the mass lesion, the logistic regression analysis demonstrated that a mass size exceeding 10 mm (OR = 2.956) and mixed mass-NME lesion (OR = 4.143) were independent risk indicators for a papilloma with high-risk or malignant lesions. For the NME lesion, the segmental or regional distribution was more commonly observed in the papilloma with high-risk or malignant lesions (61.5%) than the benign papilloma (12.5%) (P = 0.006). All the cases of occult lesions were benign papillomas.Multivariate logistic regression analysis demonstrated that clinical characteristics including being or older than 50 years (odds ratio [OR] = 4.506), having bloody nipple discharge (OR = 4.499), and concurrent breast cancer (OR = 5.083) were significant indicators for papilloma with high-risk or malignant lesions. On MRI, most papillomas presented as mass (MRI features including a mass size exceeding 10 mm, mixed mass-NME lesion, and NMEs with segmental or regional distribution indicate a papilloma with high-risk or malignant lesions. Breast intraductal papilloma is characterized by a finger-like fibrovascular core lined by epithelial and myoepithelial cells . It coulIt is a challenge for a pathologist to differentiate between benign papilloma and papilloma with high-risk or malignant lesions with the use of limited tissues obtained from core needle biopsy (CNB) , 7. CNB Studies have demonstrated that close imaging follow-up or surgical excision should be recommended for the benign papillomas diagnosed on CNB when there is a clinical or radiologic suspicious lesion , 14, 15.Mammography and ultrasound were reported not to be able to differentiate benign from malignant papillary lesions . Breast This retrospective study was approved by the Institutional Review Board of our Hospital, and the informed consent requirement was waived . Between September 2012 and August 2015, 238 patients who had intraductal papillomas confirmed by surgically excisional biopsy and preoperative MRI assessment in our hospital were retrieved from our database. The microscopic papillomas which were excised because of other breast lesions outside the papilloma and incidentally discovered by the pathologists were excluded. Among the 238 patients, the 81 patients with 87 microscopic papillomas which presented no abnormal findings on breast MRI or hardly be recognized from the background parenchymal enhancement were excluded. There was one patient who had a microscopic papilloma in one side being excluded and a papilloma in another side being enrolled. Finally, 158 patients with 175 papillomas  were enrolled.n = 56), only palpable mass (n = 54), suspicious lesions on screening ultrasound (n = 31), and other palpable mass outside the papilloma (n = 34). Preoperative Breast Imaging Reporting and Data System (BI-RADS) categories of these lesions were reviewed and the highest BI-RADS category of mammography, ultrasound, and MRI was recorded. Thirty-four of 175 lesions had a BI-RADS 3 category, and 141 lesions had a BI-RADS 4 or 5 category. Of the 34 cases who had a BI-RADS 3 category, 15 cases were diagnosed as papillomas or intraductal lesions and then biopsied, and the other 19 cases which were diagnosed as other kinds of benign lesions were also treated with biopsy instead of follow-up. All the 141 suspicious lesions with a BI-RADS 4 or 5 category were biopsied. Among the 175 lesions, 114 underwent lumpectomy, 56 underwent segmentectomy of the dilated duct, and 5 underwent quadrantectomy. Mastectomy or nipple sparing mastectomy was performed in eight patients with DCIS component after the excisional biopsy. The clinical demographics of the enrolled cases including the patients’ age, menopause status, bloody nipple discharge, personal history of papillary breast lesion and breast cancer, concurrent breast cancer , and family history of breast cancer were systematically retrieved.The indications of the 175 cases for MRI were pathological nipple discharge with or without palpable mass  was performed in 165 patients. Volume Image Breast Assessment (VIBRANT) sequence  were obtained before and at 54 s, 108 s, 162 s, 216 s, and 270 s after the intravenously injection of Gadopentetate Dimeglumine  with 0.1 mmol/kg at a flow rate of 2 mL/s and 20-mL normal saline flush.Imaging was performed on a 3.0-T whole-body MRI scanner . The patients were positioned in the prone position with both breasts placed in an eight-channel phase-array breast coil. Firstly, axial short tau inversion recovery (STIR) sequence was obtained . Next, axial diffusion-weighted imaging (DWI)  the size of the lesion was too small to identify on DWI (n = 48), or (2) DWI failure (n = 42), or (3) a cystic mass with a very thin wall to place an ROI (n = 4), or (4) occult lesions (n = 3).The size of the lesion was measured on subtracted axial images of the second series as maximum tumor diameter. The time-signal intensity curve (TIC), apparent diffusion coefficient (ADC) value, and maximum intensity projection (MIP) were obtained on a workstation . The TIC was obtained from the dynamic sequence. Oval or round regions of interest (ROIs) for obtaining TIC were set on the lesion and avoided cystic regions and duct dilation areas. According to the BI-RADS atlas , the TICP value less than 0.05 was regarded as statistically significant. According to the pathology results, the cases were classified into the group A (papilloma with or without benign proliferative lesions) and group B . Student’s t test or Mann-Whitney U test was used to compare the quantitative variables between the two groups. Chi-squared test or Fisher’s exact test was employed for the comparison of the categorical variables between the two groups. Multivariate logistic regression analysis was employed to identify clinical or MRI diagnostic risk indicators for papilloma with high-risk or malignant lesions. The variables which showed P values less than 0.1 in the univariate analysis were selected for the multivariate analysis. The “Forward: likelihood ratio” mode was adopted. The probability threshold of entry and removal for stepwise is 0.05 and 0.1, respectively.Analyses were performed by using the SPSS 23.0 (IBM Corp.) analysis software package, and a P <  0.001). Postmenopause and bloody nipple discharge were more commonly observed in group B than group A . In two patients, synchronous breast cancer was found in the same breast outside the papilloma. In six patients, breast cancer was found in the contralateral breast. In one patient, bilateral breast cancers were confirmed outside the papilloma. Concurrent breast cancer was detected more often in group B (11.6%) than group A (3.0%) (P = 0.069). No significant difference was observed in the personal histories in these two groups. Multivariate logistic regression analysis demonstrated that being or older than 50 years (odds ratio [OR] = 4.506), having bloody nipple discharge (OR = 4.499), and concurrent breast cancer (OR = 5.083) were significant indicators for papilloma with high-risk or malignant lesions , and fewer as NME  and occult lesion . Thirty cases (22.2%) of the mass lesions were papillomas with high-risk or malignant lesions. Thirteen cases (35.1%) of NME lesions were papillomas with high-risk or malignant lesions. All the three cases of occult lesions were benign papillomas, and the pathological findings revealed that the diameter of papillomas was 1 to 2 mm.On MRI, most papillomas presented as mass (P = 0.020). Papillomas more frequently presented as a solid mass (n = 121)   on MRI. Pathological findings revealed that all the cystic masses on MRI showed infarction of the papillary fronds. The presence of duct dilatation was more commonly seen in group B than group A . The mixed mass-NME lesion was also more frequently detected in group B than in group A . No significant differences were found for the location, number of masses (being solitary or multiple), shape, margin, TIC pattern, and ADC value between the two groups. Multivariate logistic regression analysis demonstrated that a mass size exceeding 10 mm (OR = 2.956) and mixed mass-NME lesion (OR = 4.143) were significant indicators for papilloma with high-risk or malignant lesions . No significant difference was found in size, duct dilation, TIC pattern, and ADC value between the two groups.The MRI features of NME lesions were summarized in Table In this study, we found patients with papillomas with high-risk or malignant lesions were older than those with benign papillomas, which was consistent with previous reports , 29. OurOn MRI, 77.1% of papillomas presented as the mass in our study. We found that a mass size exceeding 10 mm was a risk indicator for a papilloma with high-risk or malignant lesions, consistent with previous studies , 29. TomStudies have demonstrated that papillomas are usually mixed with benign proliferative lesions and less commonly with atypical and malignant lesions –9. LewisOn MRI, 21.1% cases of papilloma presented as pure NME in our study. Papilloma manifesting as NME could be due to the concomitant benign, atypical, and malignant proliferative lesions. Moritani et al.  consider2/s at b value of 1000 s/mm2, similar to that of small invasive carcinoma (1.13 ± 0.18 mm2/s) [b value of 800 s/mm2, the mean ADC value of papilloma was 1.33 mm2/s for the mass lesion. A relatively lower b value may be responsible for the slight higher ADC values of papilloma in our study. We also found that the papilloma with high-risk or malignant lesions showed similar ADC values with the benign papillomas without significant difference.Studies , 25 repoTominaga et al.  describeThe main limitation of this study is that the patients in this cohort underwent excisional biopsy instead of CNB. These patients usually had suspicious breast lesions diagnosed on mammography or ultrasound and then underwent preoperatively MRI assessment, ultrasound localization, and subsequently surgically excisional biopsy. The injection of methylene blue into the dilated ducts was performed for patients with nipple discharge before the surgical excision. Therefore, we could not assess the influence of MRI on the management of CNB diagnosed benign papilloma. Secondly, we did not compare the value of MRI with ultrasound in evaluating the extent of papillomas with pathologic correlation. Though there were limitations, we still could find some clinical and MR imaging diagnostic indicators for papillomas with high-risk or malignant lesions from this series of surgically confirmed papillomas. Our findings are applicable for patients who already had an MRI examination for a different reason, for example, screening high-risk women, or the evaluation of a suspected or a newly diagnosed breast cancer, and so on. If the patient was diagnosed as papilloma by CNB, the MRI diagnostic indicators reported in this study help to assist the surgeon for the subsequent management, surgical excision, or follow-up. A prospective study is warranted to assess the value of MRI in the management a patient with CNB diagnosed benign papilloma.In conclusion, for a patient with a papilloma, clinical features including being or older than 50 years, having bloody nipple discharge, and concurrent breast cancer were significant indicators for papillomas with high-risk or malignant lesions. On MRI, most papillomas presented as mass, and fewer as NME and occult lesion. For a papilloma, a mass size exceeding 10 mm with a mixed mass-NME pattern or NMEs with segmental or regional distribution on MRI were main indicators for a papilloma with high-risk or malignant lesions.


Herein, we present a very rare case of enteropathy-associated T-cell lymphoma (EATL) type 2 with pulmonary metastasis which was biopsy-proven. This is a very rare type of lymphoma, and no case reports or studies of enteropathy-associated T-cell lymphoma with pulmonary metastatic disease were found in the literature review.This 64-year-old male, who presented with an acute abdomen, was found to have a perforation. Subsequent pathology of the resected specimen showed neoplastic cells consistent with EATL type 2. Four months post-diagnosis, the patient developed shortness of breath. Positron emission tomography (PET) scan revealed multiple metabolically active pulmonary nodules. A biopsy of the nodules was consistent with metastatic EATL type 2 involving the lungs. Enteropathy-associated T-cell lymphoma (EATL) is a rare peripheral non-Hodgkin's T-cell lymphoma originating from intraepithelial T lymphocytes of the intestines . In genePatients typically present with abdominal pain, often associated with intestinal  obstruction, perforation, or bleeding. Other presentations include systemic/B symptoms , fatigue, infection, hepatosplenomegaly, and pruritis . The majThe patient was a 64-year-old male with a past medical history significant for hypertension, hyperlipidemia, and diabetes mellitus type 2. He presented to the emergency room with severe abdominal pain with an episode of almost passing out at home. He was in his usual state of health and noticed intermittent abdominal pain associated with nausea and vomiting for two to three weeks. He was brought to the emergency room where a computerized tomography (CT) scan of the abdomen showed a perforation involving the distal jejunum/proximal Ileum and free air. Subsequently, he underwent exploratory laparotomy and small bowel resection.The pathology of the resected specimen showed EATL type 2. The neoplastic cells had medium-sized round, darkly staining nuclei with a rim of pale cytoplasm Figure . IntraepHis postoperative course was complicated by a wound infection and delayed healing, leading to multiple antibiotic courses which resulted in the delay of subsequent care. A positron emission tomography/computed tomography (PET/CT) scan and bone marrow biopsy were done as an outpatient which showed multiple metabolically active pulmonary nodules. The largest nodule measured 2.6 cm in the posterior right lower lobe Figure . The bonThe patient subsequently presented to the hospital with shortness of breath. A CT scan of the chest showed a nodular mass-like component measuring 3.1 cm with moderate pleural effusions and multiple pulmonary nodules Figure . Biopsy Of note, he had recurrent admissions for abdominal pain where he had small bowel perforations and had surgical resections. Subsequently, he was started on chemotherapy with the CHOP regimen  once he recovered from his surgery. After he underwent the first cycle of chemotherapy, he, unfortunately, again had a perforation of the small bowel. In view of the poor prognosis, the family chose patient comfort measures only, and he succumbed to his disease.The diagnosis of the EATL is made after histopathological examination of the resected specimen since the most common presentation is a perforation. It is very difficult to diagnose the tumor on imaging studies, such as a CT scan of the abdomen, as the tumor infiltrates the wall of the intestine and does not present as a mass or growth. Even on PET scan, it is difficult to diagnose the EATL as it is more of tumor infiltration of the intestinal wall. On gross examination of the intestines, multiple circumferentially oriented ulcers are typically present and are often associated with gut perforation. There may or may not be a mass. The neoplastic cells are small to intermediate in size, with round to slightly irregular nuclei, inconspicuous nucleoli, and scant pale cytoplasm. Overall, there is a fairly monotonous appearance to the tumor cells. There is typically a sparse inflammatory background, with some cases showing scattered eosinophils.Ideally, the treatment options include chemotherapy as induction and subsequent radiation or autologous hematopoietic cell transplantation. Our patient had surgical resection as he had presented with abdominal pain due to intestinal perforation. He was subsequently started on chemotherapy with cyclophosphamide, hydroxydaunomycin, Oncovin, prednisone which, unfortunately, he could not tolerate. The prognosis of EATL is very poor because of the incidence of multifocal involvement of the small intestine and frequent dissemination. Complete resection is not achievable . MoreoveEATL type 2 is a rare disease with a very poor prognosis. Our patient presented with intestinal perforation and had evidence of disseminated disease. He was started on chemotherapy with a CHOP  regimen; unfortunately, he had an intestinal perforation after the first cycle and subsequently passed away. Treatment in these patients is a double-edged sword, as the progression of the disease itself or chemotherapy could further thin out the intestinal wall and cause perforation. Further research needs to be done on early detection of these tumors and improved treatment options to lessen morbidity and mortality.


The association between Gastrointestinal Stromal Tumours (GIST) and Neuroendocrine Tumours (NET) is very rare. These tumours have various clinical expressions and sometimes are asymptomatic. Synchronous NETs and GISTs have been already described in literature in few case reports. On the other hand, there is no mention of concomitant presence of discending colon NEC-G3 and small intestinal GIST.Presentation of case: we presented a case of a patient with clinical evidence of intestinal occlusion and radiological and intraoperative aspects of an adenocarcinoma of the left colon with a single metastasis on small bowel. The pathology analysis of the tumour showed a stenotic left colon NEC-G3 and a small bowel GIST.In this case report GIST was surgically treated as a small bowel ripetitive lesion and NET as a left colon adenocarcinoma. These tumours may have a similar presentation in terms of symptoms, endoscopic findings and imaging results.Concomitant NEC and GIST is rare but it is important to investigate patients before surgery in order to distinguish these from other tumours because of the different prognosis. Their cth an 7th decade).30–40% of GISTs develop in small bowel [Neuroendocrine tumors (NETs) are more common in the small bowel, while colorectal neuroendocrine tumors CR-NET are heterogeneous and infrequent. Colonic NETs close to the rectum are rarer and tend to be more aggressive .Their radiological identification is difficult because there are no specific signs and, most of times, only the entire bowel palpation allows to identify them .We report a case of a 62-year-old with concomitant stenotic left colon NET and small bowel GIST. This case is written according to SCARE criteria [2A 62 years old caucasian female presented to our Operative Unit complaining of severe abdominal pain in the previous 24 h and of faecaloid vomiting. The patient maintained that she did not have any flatus or bowel movements in the previous 12 h. Furthermore, she referred to have nausea and diarrhea alternated with constipation in the last year, reason why she was waiting for a colonoscopy in the following days.No previous surgery was reported in her medical history; she only had hypertension treated by beta-blockers and calcium channel blockers.Patient’s vital signs were: BP 162/100 mmHg, PF 74bpm, temperature 367 °C, RR 20, SpO2 97%, weight 64 Kg and height 166 cm.Her physical examination showed a tenderness of the abdomen, with no evidence of peritonitis, absence of bowel sounds and rectal exploration was negative.At the First Aid an ECG and an abdomen X-Ray were performed. The radiograms showed a widely dilated bowel with multiple air fluid levels.Then the patient underwent an abdomen and pelvis CT, that confirmed the dilation of the small bowel and revealed a concentric thickening (heteroplastic nature) of the walls of the first third of the descending colon. The stenosis extended for about 3,3 cm with an associated inhomogeneity of perilesional tissue , Fig. 2.Blood tests were performed with evidence of hyponatraemia (131 mmol/L) and hypocalcaemia (ionised-Ca: 4.05 mg/dL) with renal impairment , neutrophilic leukocytosis and a modest increase of C-reactive proteine (10.7 mg/L). Oncological markers at the entrance were completely negative . ASA score of patient was III.During the exploration of the abdomen, the patient had a distension of the ileal and colic loops above the stenotic left colon tract.Furthermore, an extraluminal nodule (2.3 cm), maybe a ripetitive lesion, was found on jejunal loop. It was not reported on CT. A small bowel resection of this region was performed and then a Hartmann procedure was carried out. The operation lasted 190 min.Post operatively, the patient had a protract hypotensive state .There were no complications of surgical interest. On postoperative (POD) day 2, the patient presented a temperature of 382 °C and she was treated by antipyretics. An urine culture test was performed with evidence of Enterococcus faecalis. Then a targeted antibiotic therapy was administered.Diet was advanced as tolerated and patient was discharged on POD 17 after achieving an adequate nutritional status and without any antihypertensive therapy.The pathology analysis revealed a poorly differentiated neuroendocrine adenocarcinoma (NEC-G3) of the discending colon with endovascular invasion and full-thickness infiltration of the muscular wall, involving the pericolonic adipose tissue with 13 metastatic lymph nodes (pT3N2b).The jejunal nodule, suspected of repetitive lesion, was a low-grade recurrence GIST.The immunophenotype analysis showed that NEC was CK20 +, Sinaptophysin +, and focally positive for CD56 (negative for CK7 and Chromogranine) with an Ki67 of 80%. GIST was positive for CD117, CD34 and negative for Smooth Muscle Actin (SMA) with mitotic counts of <5/50 hpf. , Fig. 4FAt the discharge, Neuron Specific Enolase (NSE) and Chromogranin A were dosed. They resulted negatives .On POD 22, the wound healing process was normal without local complications. Furthermore, the patient reported a normalization of blood pressure values and started chemotherapy.3The coexistence between GISTs and other neoplasms is pretty rare, ranging from 4.5 to 33% of cases .Gastrointestinal stromal tumours (GISTs) are infrequent, generally solitary lesions with an incidence of around 1/100 000 per year. The most common site of this tumour is stomach (60%), followed by small intestine (30%), colon–rectum (5%), and esophagus (1%). A small percentage of GISTs has been described in the omentum and mesentery (4%) .Usually their surgical treatment consists of simple enucleation or segmental resection.Neuroendocrine gastroenteropancreatic tumours (GEP-NETs) are a heterogeneous group of tumours with their origin in neuroendocrine cells of the embryological gut. Most commonly, the primary lesion is located in the gastric mucosa, the small intestine or pancreas. Colorectal location is uncommon but its rate of detection is increasing thanks to the widespread use of colonoscopy.Rectal neuroendocrine tumors are more frequent (137% of NET), usually small lesions and their histological malignancy is low to moderate , whereas colon NET are rarer , aggressive, poorly differentiated and more malignant (G3) .Colorectal NETs have non-specific symptoms such as obstruction, hematochezia, pain, weight loss and changes in bowel habits .Given that most of colonic NETs are right sided, they may grow considerably before symptoms appear. Rectal and left colon NETs may cause constipation, bleeding , tenesmus and, sometimes, pain .Carcinoid syndrome, consisting of flushing, diarrhea, nausea, and cardiac symptoms is very uncommon. Less than 5% of patients with colorectal NETs develops these symptoms .Histological type of neoplasm according to WHO classification, histological grading and immunohistochemical tests   are impoTreatment of colonic NETs is similar to colonic adenocarcinoma, with segmental resection and lymphadenectomy .Synchronous NETs and GISTs have already been described in literature in few case reports . On the In this case the patient had a stenotic left colon tumour (reported on CT), but during the exploration of the abdomen a nodular extraluminal lesion was found on an intestinal loop. The surgical approach, considering the patient’s health state and the suspected ripetitive lesion, was the Hartmann porcedure and a segmental resection of the small intestine, in order to allow a rapid start of an adjuvant treatment after surgery.In fact, the colostomy began to work on POD 2 and the patient underwent a FOLFOX regimen of chemotherapy 20 days after the discharge.4Since the concomitant presence of GIST and NET is rare, it is hard to suspect their association in a case like the one we presented. However, its surgical approach does not change. In fact, in this case GIST was treated as a small bowel ripetitive lesion and NET as a left colon adenocarcinoma.Although they may have an expression similar to the more common metastatic adenocarcinoma in terms of symptoms, endoscopic findings and imaging results, it is important to distinguish them because of their different prognosis.Colonoscopy is always suggested when there is a change in the bowel habitus, since NET can be endoscopically treated when small ,17. EvenEarly diagnosis of concomitant tumors such as GIST and NET can guarantee a better prognosis of patients.No conflicts of interest to declaire.No funding received.All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Amoruso Michele: first surgeon, conception of study design, data collection, analysis, manuscript writing, revision and manuscript submission.Papagni Vincenzo: conception of study design, data collection, analysis, manuscript writing and revision.Picciariello Arcangelo: conception of study design, data collection, analysis, manuscript writing and revision.Pinto Vito Leonardo: critical revision of the manuscript, approved the final version of the manuscript for submission.D’Abbicco Dario: conception of study design, data collection, analysis, manuscript writing and revision.Margari Antonio: second surgeon, drafting, revising of the manuscript and participation in the care of the patient.N/A.Michele Amoruso MD.Not commissioned, externally peer reviewed.


Following thorough investigation with imaging and Tru-cut biopsy, finally, an excisional biopsy was done. Histopathological examination confirmed a desmoid tumor, and the patient received adjuvant radiotherapy. At the one-year postoperative follow-up, there was no recurrence; the patient had been explained the prognosis. This case highlights a rare site of an extra-abdominal desmoid but with classical clinical presentation, imaging, intraoperative, and histopathological findings. Awareness and knowledge of this entity are of paramount importance for clinical practitioners.Desmoids are rare soft-tissue tumors of the abdominal wall that may sporadically occur extra-abdominally. It manifests as clonal fibroblastic proliferation with an infiltrative tendency and capacity to recur without metastasizing. An adolescent male presented with a gradually increasing globular, non-tender, firm, non-pulsatile swelling (8 × 5 × 3 cm Muller first coined the term desmoids; these tumors have been identified to arise from fascial and musculo-aponeurotic tissue, mostly in the abdominal wall . However3 and was situated in the left popliteal fossa since five months  of the lesion revealed an ill-defined, lobulated, soft-tissue, space-occupying lesion in the lateral aspect of the calf adjacent to the lateral head of the gastrocnemius that was hypointense on the T1-weighted image and hyperintense on the T2-weighted image, with multiple ill-defined T2-weighted signal voids within the lesion that gave an impression of hemangioma Figures -2C.Tru-cut biopsy showed spindle cells arranged in clusters and scattered singly in a hemorrhagic background with no evidence of malignancy, suggesting a benign spindle cell tumor. Histopathological examination with Hematoxylin–Eosin staining revealed plenty of fibroblasts spread against a background of collagen as well as infiltration of the adjoining healthy tissue that led to a microscopic diagnosis of desmoid fibromatosis Figure .Blood investigations as part of the routine preoperative workup showed normal results. Clinically, desmoid tumor mimics other soft-tissue tumors such as leiomyoma, rhabdomyoma, nerve sheath tumor, as well as vascular and perivascular tumors. Common histological differential diagnoses include fibrosarcoma, spindle cell tumor, epithelioid tumor, and pleomorphic tumor.Under general anesthesia, the patient was positioned prone and a J-shaped lazy posterior incision was made. The tumor was situated deep to the fascia although it involved it. On palpation, it was firm, partly smooth, and seemed to arise from the deep fascia and muscle without any clearly defined planes; it was located adjacent to the lateral head of the gastrocnemius. The mass did not engulf any major vascular structures in the popliteal fossa although the common peroneal nerve and the medial cutaneous sural nerve traversed it. The sural nerve was sacrificed and the common peroneal dissected out carefully; the mass was then removed, and the specimen in toto was sent for histopathological study Figure .The wound was closed in layers. There were no immediate postoperative complications. The dorsiflexors of the ankle and toes were well preserved. The patient was discharged on tenth postoperative day and received adjuvant radiotherapy after three weeks to minimize any chance of recurrence. At the one-year follow-up, the patient had no radiological or clinical evidence of recurrence, and has been advised regular follow-ups every six months.With an incidence of approximately three to four cases per million, desmoids comprise only 3% of all soft-tissue tumors . The typThe popliteal fossa is an unusual location for an extra-abdominal desmoid-type fibromatosis. Diagnosing this tumor is challenging and histopathological study is necessary for a final diagnosis. Adjuvant radiotherapy minimizes chances of tumor recurrence.


Ingestion of foreign bodies can cause various gastrointestinal tract complications including abscess formation, bowel obstruction, fistulae, haemorrhage, and perforation. While these foreign body-related complications can occur in normal bowel, diseased bowel from inflammation, strictures, or malignancy can cause diagnostic difficulties. Endoscopy is useful in visualising the bowel from within, providing views of the mucosa and malignancies arising from here, but its ability in diagnosing extramural malignancies arising beyond or external to the mucosa of the bowel as in the case of metastatic extramural disease can be limited.  We present the case of a 60-year-old female with an impacted chicken bone in the sigmoid colon with formation of a sigmoid mass, on a background of metastatic lung cancer. On initial diagnosis of her lung cancer, there was mild Positron Emission Tomography (PET) avidity in the sigmoid colon which had been evaluated earlier in the year with a colonoscopy with findings of diverticular disease. Subsequent computed tomography (CT) scans demonstrated thickening of the sigmoid colon with a structure consistent with a foreign body distal to this colonic thickening. A repeat PET scan revealed an intensely fluorodeoxyglucose (FDG) avid mass in the sigmoid colon which was thought to be inflammatory. She was admitted for a flexible sigmoidoscopy and removal of the foreign body which was an impacted chicken bone. She had a fall and suffered a fractured hip. During her admission for her hip fracture, she had an exacerbation of her abdominal pain. She developed a large bowel obstruction, requiring laparotomy and Hartmann's procedure to resect the sigmoid mass. Histopathology confirmed metastatic lung cancer to the sigmoid colon.  This unusual presentation highlights the challenges of diagnosing ingested foreign bodies in patients with metastatic disease. Around 20% of ingested foreign bodies fail to pass through the gastrointestinal tract . These cOur patient had an impacted chicken bone in the sigmoid colon in the setting of metastatic non-small-cell lung cancer. This was investigated radiologically and found to be an intensely FDG-PET avid mass, initially presumed to be either an inflammatory mass related to the chicken bone impaction or metastatic disease related to her lung cancer. This mass appeared to resolve upon removal of the chicken bone; however, she represented later with a subacute large bowel obstruction related to the sigmoid mass which was found to be metastatic lung cancer at surgery. Consequently, our case highlights the difficulties of establishing a diagnosis in this complex case.In this case report, we present a literature review of colonic chicken bones and investigate similar patterns across the various presentations reported. PubMed and Google Scholar were both utilised to identify the search terms “chicken bone” AND “bowel” OR “large bowel” OR “colon”. The results were systematically reviewed to include only case reports of chicken bones in the large bowel, while the details of each case were analysed for the purposes of the literature review.We present the case of a 60-year-old lady who initially presented with a pseudomonas empyema and a right hilar mass. Initial diagnostic bronchoscopy revealed no endobronchial lesion. She was treated under the respiratory and infectious diseases' teams with decortication and antibiotics which resulted in marked clinical improvement. Follow-up imaging showed a persistent right hilar mass, necessitating a repeat diagnostic bronchoscopy and biopsy. This revealed a non-small-cell lung cancer (NSCLC) adenocarcinoma which was EGFR and ALK negative.Baseline staging imaging revealed that she had metastatic disease with a right lung primary lesion, mediastinal nodes, and adrenal, frontal skull bone, and left pelvic bone metastases (T4N2M1c). She underwent an FDG-PET scan as part of her staging investigations in June 2017, revealing an area of intense heterogenous FDG-PET avidity in the sigmoid colon. This was suspicious for a metastatic deposit or a complication secondary to diverticular disease . HoweverThe patient had minimal comorbidities and palliative systemic treatment, including radiation, was organised. She proceeded to carboplatin plus gemcitabine chemotherapy and completed 4 cycles in September 2017. She received palliative radiation to the right frontal bone and left pelvis metastatic deposits. She was then commenced on maintenance pemetrexed chemotherapy in October 2017.In March 2018, she had a repeat colonoscopy, which revealed two polyps and evidence of diverticulosis in the sigmoid and descending colon. The polyps were removed, and histopathology revealed no evidence of malignancy.In April 2018, she developed asymptomatic low-volume brain metastases in the left temporal, left occipital, and right posterior frontal lobes ranging from 3 mm to 16 mm in diameter. She underwent gamma knife treatment to these lesions and proceeded to Nivolumab immunotherapy in April 2018.After 2 cycles of Nivolumab, our patient developed mild lower abdominal pain, which she complained of during her outpatient oncology visits. This had been diagnosed as diverticulitis by her general practitioner, who commenced antibiotic treatment.A CT scan demonstrated circumferential thickening of the bowel wall in the sigmoid colon and a suspicious-looking intraluminal tubular structure distal to this, suspicious for a foreign body Figures  and 4. TThe patient continued to eat normally during this time and reported no changes in her bowel habits. She had no fevers and the only abnormality on her blood results was a raised C-reactive protein. The clinical decision was to follow this closely with serial imaging. Progress imaging 2 weeks later confirmed persistence of this foreign body. Consequently, our patient was admitted due to ongoing lower abdominal and suprapubic pain and for intravenous antibiotics. A repeat FDG-PET-CT scan was conducted, revealing an intensely FDG avid mass in the midsigmoid colon . The incDespite these findings, it was still possible that this was secondary to an inflammatory rather than a neoplastic process. The patient was scheduled for a flexible sigmoidoscopy to evaluate the intracolonic foreign body. This revealed a chicken bone impacted in the sigmoid colon . The extThe patient unfortunately represented the day after discharge with a hip fracture following a mechanical fall. She underwent a hip replacement and during her postoperative recovery developed more abdominal pain. A further CT scan raised concern that this mass had become an intramural abscess with images displaying some gas locules within it . She wasShe was discharged and remained well the first week following her discharge. The following week, she developed worsening pain, fevers, and a subacute large bowel obstruction. She underwent an emergency laparotomy, at which time, she was found to have a large, fungating, and hard mass, which was densely adherent to the bladder. She underwent a resection of this sigmoid mass along with a contiguous segment of the bladder . The segUnfortunately, during the course of her recovery, our patient had another fall and broke her other hip. She has since had this hip replaced and has recovered from her surgery and is managing her stoma. She underwent further rehabilitation and was discharged home. She remains on systemic treatment for metastatic lung cancer.Our case represents a rare and unusual presentation of an impacted chicken bone in the setting of a sigmoid mass. Thirty-six reports of complications as a result of chicken bones in the large bowel were identified in the English literature . The sigIn our review of the 36 reported cases of complications from chicken bone ingestion, nonspecific abdominal pain was a common presenting complaint throughout, in similar fashion to how our patient presented. Radiology formed a cornerstone in the workup of patients with ingested foreign bodies, with CT and X-ray of the abdomen standing out as the most common investigations organised. Ultimately, endoscopy served as the most common means of gaining a definitive diagnosis, while concomitantly managing the condition. Surgery, however, was necessary in cases where the chicken bone had led to serious complications.In terms of these complications from ingested chicken bones, bowel perforation was noted to occur in 19 of the 36 case reports analysed. A history of gastrointestinal disease, such as diverticulosis and colonic malignancy, predisposes individuals to experiencing complications of ingested foreign bodies, especially that of perforation [A review of the literature revealed that patients with a history of alcoholism, dentures, or sensory neuropathy are most at risk of swallowing a foreign body . Our patOur patient was on an immune checkpoint inhibitor for her lung cancer. Immune checkpoint inhibitors have known immune-related gastrointestinal toxicities such as diarrhoea and colitis. Rare cases of bowel perforation requiring colostomy have been reported in the literature . The devTo our knowledge, this is the first case of chicken bone impaction in the setting of metastatic lung cancer to the sigmoid colon. Some of the difficulties, even with modern imaging and FDG-PET, in differentiating inflammatory from neoplastic processes in the bowel are described. This case highlights that while colonoscopy is useful in visualising the bowel from within and is crucial in diagnosing malignancies arising from the bowel mucosa, its ability in diagnosing extramural malignancies arising beyond or external to the mucosa of the bowel as in the case of metastatic extramural disease can be limited.Foreign bodies mostly present with nonspecific abdominal pain, and while the majority are managed surgically, they can sometimes be retrieved endoscopically. In the large bowel, the sigmoid colon is the most common site of complications arising from ingested chicken bones. The literature review identified that perforation of the bowel tends to occur in the setting of diverticular disease and malignancy. Our case reflects the diagnostic complexity in a patient with an ingested foreign body in the setting of metastatic disease, despite modern radiological investigative modalities and endoscopy. This report highlights the value of keeping ingested foreign bodies in mind when formulating differential diagnoses for nonspecific abdominal pain. At the same time, it identifies a key area in oncological practice, where rigorous follow-up is essential to the screening for metastasis of primary malignancies to distant organ sites.


Compressive myelopathy and compression fracture of aggressive vertebral hemangioma after parturition is a rare condition. Vertebral body compression fracture and high serum progesterone lead to extraosseous hemangioma enlargment cause narrowing the spinal canal which contribute to compressive myelopathy relate to pregnancy.We report a case of compressive myelopathy and compression fracture of aggressive vertebral hemangioma after parturition in a 35-year-old woman. The patient complained unable to walk and experienced intense pain in the back.Based on the clinical features and imaging studies, the patient underwent a T4–T6 laminectomy. Histopathology consistent with vertebral hemangioma.The patient underwent laminectomy for decompression. After subperiosteal dissection of the paraspinal muscles and exposure of the laminae, there was no involvement of the lamina by the tumor. The epidural tumor was removed through the spaces lateral to the thecal sac. Vertebroplasty was performed through T5 pedicles bilaterally and 7 ml of polymethylmethacrylate (PMMA) cement was injected. T4–T6 pedicle screw fixation was performed for segmental fixation and fusion.Six months after resection of the tumor the patient remained asymptomatic. She reported no low back pain and had returned to her normal daily activities, with no radiographic evidence of recurrence on MRI. Physical examination revealed that superficial and deep sensation was restored to normal levels in the lower extremities.The occurrence of compressive myelopathy of pregnancy related vertebral hemangiomas is quite unusual. It can lead to serious neurologic deficits if not treated immediately. So, prompt diagnosis is important in planning optimal therapy and preventing morbidity for patients. Pregnancy can exacerbate the growth of hemangiomas, resulting in spinal cord compression.–6 Compressive myelopathy related to pregnancies of vertebral hemangiomas may occurred during gestation or postpartum and there were 33 cases had reported in the literatures. Most common clinical presentation of reported patients were paraplegia. We herein report a rare case of an aggressive vertebral hemangioma that caused a compression fracture and compressive myelopathy 4 days after vaginal delivery. There were 6 cases aggressive vertebral hemangioma caused compressive myelopathy post-parturition and the patient we reported was the first case that occurred after delivered the second baby. The current case was aggressive vertebral hemangiomas and the component extended to epidural space shows no uptake in PET-CT which may differentiated from neurinoma, meningioma, and lymphoma. We also reviewed literatures and analyzed compressive myelopathy related to pregnancies.Vertebral hemangiomas are the most common benign tumors of the spine and have a prevalence of 10% to 12% of the general population.2A 35-year-old woman with no relevant medical history was admitted to our hospital with a 15-day history of progressive numbness and weakness of her bilateral lower extremities that began 4 days after vaginal delivery. She had given birth to a child 5 years before and the history was unremarkable. After admitted, the patient was unable to walk and experienced intense pain in the back, especially during mobilization. Physical examination showed percussion pain at the T4 to T8 level. Neurological examination revealed decreased muscle strength in the bilateral lower limbs. Her muscle strength was grade 3 in both lower extremities. Sensory examination showed impaired temperature and pain sensations with a sensory level at T6. The Babinski sign was positive bilaterally.Computed tomography (CT) showed sclerosis of the cancellous bone involving the entire T5 vertebra with epidural spaces extension, including the epidural space Fig. A. ConvenBased on the clinical features and imaging studies, the patient underwent a T4–T6 laminectomy. After subperiosteal dissection of the paraspinal muscles and exposure of the laminae, there was no involvement of the lamina by the tumor. A firm epidural mass was found filling the anterior epidural space and compressing the cord. The epidural tumor was removed through the spaces lateral to the thecal sac. The dura was found to be normal with no evidence of infiltration from the tumor. Vertebroplasty was performed through T5 pedicles bilaterally and 7 ml of polymethylmethacrylate (PMMA) cement was injected Fig. A-B. T4–THer symptoms were rapidly relieved after the surgery. Postoperative plain x-ray showed a good alignment of the thoracic spine and stable fusion of T4-T6. Six months after resection of the tumor the patient remained asymptomatic, with no clinical or radiographic evidence of recurrence on MRI Fig. C-D. Phys3–9 The typical radiological characteristic of vertebral hemangiomas is vertical striation sproduced by zones of reduced bone density between more dense trabeculae.,8,10 The lesion may involve the pedicles, arches, and spinous processes. The vast majority of these lesions are asymptomatic.Vertebral hemangiomas are common benign vascular tumors. The most frequent location of these lesions is within the lower thoracic and lumbar vertebrae, and they are often multiple. The affected vertebra is usually located between T3 and T8. Vertebral hemangiomas can cause neurologic symptoms by means of various mechanisms. For example, enlargement of the vertebral body leads to narrowing and distortion of the spinal canal and a symptomatic compression fracture.The term “aggressive hemangioma” refers to a vertebral hemangioma with extraosseous extension. Pathophysiologically, vertebral hemangiomas cause diffuse bone infiltration and consequently reduced bone density. The lesions are characterized by erosion of the horizontal trabeculae. Rarely, their enlargement within the vertebral body is weakened by the hemangioma. These 2factors are responsible for pathological fracture under axial loading.Compression fracture of an involved vertebra is infrequent because the hemangiomatous vertebra is reinforced by thick sclerotic vertical trabeculae from new bone formation.There were 33 cases vertebral hemangiomas, including present case, with compressive myelopathy related to pregnancies reported in the literatures which listed in Table Pregnancy is known to provoke progression of a vertebral hemangioma by inducing a 30% to 50% increase in blood volume. The 2 main mechanisms for dilatation or expansion of hemangiomas during pregnancy are as follows:1.the gravid uterus may compress the inferior vena cava, resulting in a venous pressure increasein the paravertebral venous plexus, and2.,12hormonal changes such as high serum progesterone may lead to expansion of the hemangioma and development of neurological symptoms. In the reported compressive myelopathy related to pregnancies, 2 case caused compressive myelopathy because of bulging of the vertebrae which reported by Askenasy et al and Redekop et al respectively,6 cases with vertebral compression fracture. Among these 6 patients, 2 cases were aggressive hemangioma which may aggravate clinical symptom.In this condition, mechanical factors such asbending and axial vertebral loading lead to the development of a pathological compression fracture. reported the first case of a symptomatic vertebral hemangioma during pregnancy in 1957. Among the reported 33 case compressive myelopathy related to pregnancies, 6 cases were post parturition. The present case is unusual because neurological deterioration did not begin until after parturition, and her symptom occurred 4 days after vaginal delivery. Staikou et al and Schwartz et al reported aggressive vertebral hemangiomas with compression fractures with immediate development of compressive myelopathy after vaginal delivery, and the imaging findings are similar to present case. Two cases of compressive myelopathy without compression fracture were reported by Oppenlander et al and Tekkök et al, respectively. The specific time at which the myelopathy occurred in the report by Tekkök et al was unavailable, unlike present case, extradural component of hemangioma located at the posterior aspect of the spinal cord. The myelopathy reported by Oppenlander et al occurred 4 months after the patient gave birth, in this patient, extradural component of the hemangioma show big mass which located intervertebral foramen and paraspinal space.Askenasy et alThe characteristic MRI appearance of a vertebral hemangioma involves T1 and T2 hyperintensity on non-contrast imaging. The extraosseous component is hypointense relative to the marrow on T1-weighted imaging and hyperintense on T2-weighted imaging and shows uniform enhancement on post-enhanced images. 18-FDG PET/CT is helpful for determining the extent of the lesions. The mild uptake in the T5 vertebra may be caused by a compression fracture; however, vertebral hemangiomas without a compression fracture also may show mild FDG uptake. The lack of uptake by an extraosseous component may help to differentiate the lesion from other extradural space-occupying lesions such as hemangioblastoma, schwannoma, ganglioglioma, and metastatic tumors, which usually show moderate FDG uptake.,17 In these cases, preoperative embolization procedure is an effective method which can decrease the risk of hemorrhage. Most of the reported 33 patients performed laminectomy and the postoperative course was uneventful with complete neurological recovery.The rapidly increasing size of hemangiomas during pregnancy can cause narrowing of the spinal canal, fracture of the involved vertebral body, causing acute neurological deficit. Patients will warrant emergency surgical decompression.4 The differential diagnosis of aggressive vertebral hemangioma includes nerve sheath tumor, especially extradural component located in the intraspinal space, intervertebral foramen, and paraspinal area, which are dumbbell-shaped. The involved vertebral bodies and elements of hemangioma manifest as thick trabeculae on CT images and the extradural components show homogeneous hyperintense on T2-weighted images, which is the imaging characteristics of aggressive vertebral hemangioma.The occurrence of compressive myelopathy of pregnancy related vertebral hemangiomas is quite unusual. It can lead to serious neurologic deficits if not treated immediately. So, prompt diagnosis is important in planning optimal therapy and preventing morbidity for patients.www.liwenbianji.cn/ac), for editing the English text of a draft of this manuscript.We thank Angela Morben, DVM, ELS, from Liwen Bianji, Edanz Editing China (Investigation: Guanxi Wang, Yuandong Mu, Junyi Che.Writing – original draft: Junyi Che, Guangfei Zhang, Gang Jiang.Writing – review & editing: Guanxi Wang, Chuan-ping Gao.


Prepubertal primary testicular tumors account for ∼1% of all pediatric solid tumors. We report a new case of prenatal diagnosis of juvenile-type granulosa cell tumor (JGCT). A fetal ultrasound performed at the 38th week of gestation for suspected nonvertex presentation identified a left multilocular septated cystic testicular mass, suggestive for JGCT. At birth, a painless left scrotal mass was detected. Ultrasound re-evaluation excluded torsion of the testis. Tumor markers and abdominal ultrasound were normal for age. Inguinal exploration revealed a cystic mass beneath the tunica albuginea that had replaced all the normal parenchyma. Since organ-sparing surgery was thus not feasible, an orchiectomy was performed and diagnosis of JGCT was confirmed. At 7-year follow-up, the child presented an uneventful outcome. Our case shows that neonatal JGCT, which has an intrauterine genesis, can be diagnosed prenatally by ultrasound in the last weeks of pregnancy. Juvenile-type granulosa cell tumor (JGCT) of the testis, although rare, is the most common testicular neoplasm in the first 6 months of life.A healthy 2-day-old newborn was admitted to our department for a left scrotal mass. He had undergone a prenatal ultrasound a week before delivery (38th week of gestation) for suspected nonvertex presentation. On that occasion, a left testicular cystic mass (2 × 2 cm), suspected to be a JGCT due to its multicystic aspect, was identified. At clinical presentation, the left testicle appeared in situ, with increased size and consistency, while the right testicle and the penis were normal.Ultrasound revaluation after birth excluded torsion of the testis and confirmed the presence of a voluminous multicystic left testicular mass without normal-appearing parenchyma. Serum α-fetoprotein (AFP) and β-human chorionic gonadotropin (β-HCG) were normal for age. Karyotype was normal. Abdominal ultrasound did not show any anomalies. Following surgical oncological criteria, we opted for an inguinal approach: after groin incision, the spermatic chord was identified and clamped at the level of the deep inguinal orifice. Testis examination revealed a cystic mass beneath the tunica albuginea replacing all normal parenchyma. We performed funiculo-orchiectomy, since organ-sparing excision of the mass was considered not possible. There was no evidence of enlarged inguinal lymph nodes.Gross examination of the surgical specimen revealed a well-circumscribed 2 × 1.5 cm white mass. The cut surface showed multiple, thin-walled cysts containing clear fluid .Microscopic examination showed multiple follicle-like structures of varying size, round to oval, filled with basophilic fluid stained by mucicarmine. The follicles were lined by variable layers of cells with round hyperchromatic nuclei and scant pale cytoplasm . NucleoBased on morphologic and immunohistochemical findings, a diagnosis of JGCT was formulated. The tumor did not extend into the spermatic cord, epididymis, or tunica vaginalis. Nevertheless, only a small rim of residual testis was present.The baby was discharged 2 days after the surgical procedure. Seven-year-follow-up was uneventful.Prepubertal primary testicular tumors account for ∼1% of all solid pediatric tumors. Gonadal stromal tumors, including Leydig cell, Sertoli cell, and granulosa cell tumors, account for ∼8% of these neoplasms and are therefore extremely rare. JGCT accounts for only 1.2% of all prepubertal testis tumors recorded in the Pre-pubertal Testis Tumor Registry.Differential diagnosis for JGCT includes evaluation for yolk-sac and other sex cord-stromal tumors.Overall, the typically very young age of patients is helpful in diagnosisIn one case reported in the literature, retrospectively reviewed prenatal ultrasounds of a newborn treated for JGCT showed a testicular mass.Due to their benignancy, JGCTs have been treated with testis-sparing surgeryOur case shows that JGCT can be diagnosed prenatally in the last weeks of gestation. In our case, diagnosis in the 38th weeks of pregnancy after unremarkable previous routine ultrasounds indicates late fetal development of the tumor. Although successful trans-scrotal removal of JGCT has been reported, inguinal orchiectomy remains the safest way in terms of oncological criteria.


The patient initially presented with abdominal pain, and baseline imaging demonstrated several hepatic adenomas, the largest of which (approximately 8 cm) was adjacent to the inferior vena cava. Owing to the location of this adenoma, surgical/vascular intervention was deemed inappropriate. The patient was actively observed for approximately 4 years, and managed supportively during any recurrent episodes. With follow-up CT/MRI scans, the “natural history” of this particular lesion, including haemorrhage, thrombosis and infarction, was observed. However, as intervention was unsuitable, further MRI was performed in view of these complications, allowing observation of the end-stage features of the adenoma. Appearances were consistent with a rare complication of hepatic adenoma,  HB, a 33-year-old female, first presented in November 2012 with severe right upper quadrant pain. She was on the oral contraceptive pill at the time. Baseline ultrasound, CT and MRI scans demonstrated multiple liver lesions with features in keeping with hepatic adenomata. The patient re-presented 4 weeks later, and repeat imaging revealed likely infarction of the largest lesion.Owing to the location of the dominant adenoma, resection/intervention was not advisable and HB was closely monitored by the regional liver unit and local hepatology team.Over the next few months, the patient generally improved and follow-up MRI was performed at 10 months from initial presentation, which demonstrated slight involution of the infarcted adenoma.For the next 3 years, she was followed up annually by the local hepatology team.In August 2016, an episode of acute epigastric pain with newly deranged liver biochemistry prompted a review by the local hepatologist and an updated MRI.Fibrolamellar carcinomaHepatocellular carcinomaMetastasesT2 fat-saturated MRI the lesions were isointense and diffusion-weighted imaging was suggestive of benign aetiology; dynamic sequences showed diffuse arterial enhancement and prompt washout with pseudocapsule appearance  and features in keeping with infarction.Interval follow-up MRI during the periods of April 2013 to September 2015 showed essentially stable disease, with slight involution of the infarcted adenoma .A single phase  CT was performed when the patient presented with an acute episode of pain in August 2016 while on holiday, which demonstrated an increase in size and heterogeneous enhancement of the previously infarcted adenoma suggestive of recent haemorrhage or further infarction. Follow-up MRI , 4 perfoSupportive management including antibiotics was given (there were concerns about superadded infection following haemorrhage).Following discussion with the regional liver unit and the local team, the largest adenoma was still not felt amenable to intervention and the patient is to be managed with active observation.1 Unfortunately, in the case of HB tissue samples were not available to facilitate classification, owing to the location of the lesion.Hepatic adenomas are benign tumours of the liver, derived from the monoclonal proliferation of hepatocytes. There are four molecular pathological subtypes of hepatic adenoma (HA). These are (1) HAs with inactivating mutations of hepatocyte nuclear factor 1α , (2) HAs with activating mutations of β-catenin gene (HA-B), (3) HAs without mutations of the HNF1A or β-catenin genes and with inflammatory features (HA-I) and (4) unclassified HAs that have no specific gene mutations or unique morphologic features (HA-U).3 Typically, in an uncomplicated hepatic adenoma, MR imaging demonstrates a well-defined isointense or slightly hyperintense lesion on T2 weighted images and heterogeneous signal on T1 weighted images.4 With gadolinium, the lesion enhances markedly and homogeneously during arterial phase with complete washout during the portal phase.5 Approximately 4–7% of adenomata contain fat, which is echogenic on ultrasound (mimicking haemangioma), low attenuation on CT and high signal on T1 weighted images.MR imaging appearances of HAs are variable and non- specific.5 These are well documented and described in textbooks and literature. A very rarely described phenomenon is that of cystic degeneration.6Complications arise when the adenomata outgrow their blood supply and can include rupture, haemorrhage, thrombosis, infarction and malignant transformation.7 but this requires strict patient selection criteria. Based on these criteria, HB would have been an unsuitable candidate because of the size of the largest adenoma and its location adjacent to the inferior vena cava.8 Resection of adenomatous disease is further contraindicated by the presence of concurrent background fatty liver and multifocality.Conventionally, adenomatous disease is surgically resected owing to the rare potential for malignant transformation and risk of the above complications. Recent literature suggests the possibility of treatment with radiofrequency ablation (RFA),9Transarterial embolization (TAE) has not been prospectively investigated as a means of treatment for cases where surgical resection and RFA are contraindicated. Research published in 2009 indicated that watchful waiting with TAE during episodes of haemorrhagic complication may be the best treatment option for this group of patients at present.The initial MR imaging of HB revealed liver lesions consistent with hepatic adenomas, and later demonstrated some of the more conventional sequelae including haemorrhage and infarction. However, the most recent MR imaging had features consistent with a rarely described consequence of haemorrhagic/infarcted hepatic adenoma—cystic degeneration.e.g. uterus, was therefore performed. Cystic degeneration is a form of degeneration that occurs following oedema, e.g. following haemorrhage and necrosis. The regions of oedema result in the formation of cysts with walls.10 Key features of uterine cystic degeneration include high signal intensity on T2 weighted images  that do not enhance with gadolinium.11 These appearances were demonstrated on HB’s most recent MRI, and although rare, would be explained by a process of cystic degeneration following the haemorrhage/infarction of a large hepatic adenoma.The MRI features of cystic degeneration in hepatic adenomas are not well documented and scarcely found on searching the literature. A search for cystic degeneration of tumours in other anatomical locations, Imaging characteristics are different for different subtypes of hepatic adenoma.Haemorrhage or infarction of hepatic adenomas can result in the rare complication of cystic degeneration. MRI features appear similar to cystic degeneration elsewhere.Surgical resection and radiofrequency ablation remain the mainstay of treatment for hepatic adenoma provided the patient meets the selection criteria.Written informed consent for the case to be published  was obtained from the patient for publication of this case report, including accompanying images.


In this study, we report a rare case of osteoma cutis (OC) and tonsillolith, diagnosed using cone beam computed tomography. The dystrophic calcifications in the face and tonsils were incidentally found during examination of the patient’s scan with no relation to the main chief complaint. The diagnosis was OC, combined with dystrophic calcification of the tonsils. It is important to mention that OC is a rare soft-tissue ossification of cutaneous tissue, typically on the face and clinically asymptomatic. It may be primary but the majority of cases are secondary. Incidental finding of OC and tonsilloliths on a two-dimensional dental radiograph does not provide sufficient information concerning the location of these calcifications. Thus, cone beam computed tomography (CBCT) provides critical information for the diagnosis of asymptomatic OC lesions not available through any other means of clinical detection. Osteoma cutis (OC) and tonsilloliths are soft-tissue calcifications in the head and neck region that result from the deposition of minerals at specific sites and may be of pathological, age-related or idiopathic origins . OC is aAnother example of soft-tissue calcification is tonsillar stones or tonsilloliths. These lesions occur as white or yellow densifications in tonsillar crypts that originate from microorganism and tissue debris retention in the crypts of palatine tonsils . TonsillIn dentistry, routinely performed dental radiographs can capture the lesion most commonly in the lips and cheek regions; however, due to difficulty in localization, interpretation of OC cases has always been challenging especially using conventional imaging. CBCT, with its 3D software based volumetric reconstruction and multiplanar evaluation with a distortion-free image, offers an important clinical tool facilitating detection and diagnosis of these calcific lesions . Cone beThe present case reports OC in a combination of tonsillar stones. This condition was an incidental finding identified during a CBCT scan evaluation, apart from the main chief complaint of the patient without related symptoms. The purpose of this presentation is to underscore the importance of CBCT for evaluating OC lesions by the dental professional using three-dimensional radiography.A 33-year-old male presented to be admitted as a patient at Case Western Reserve University School of Dental Medicine clinic in Cleveland, Ohio for the purpose of obtaining dental implants. However, the patient complained of pain in his upper jaw bilaterally. His medical history was free from systemic diseases but indicated a past history of acne and recurrent tonsillitis. His vital signs were recorded as 126/85 mmHg blood pressure, pulse of 103 bpm, 15 respirations per minute, height 6.1 ft, weight 196 lb, a calculated body mass index (BMI) of 23.71. No other medical conditions were listed, and the patient did not report taking any medications.Upon intraoral examination, badly decayed upper right second molar and highly restored left second molar were noticed with pain on percussion. Dental periapical lesions were suspected. The patient was referred to a private dental imaging center for imaging of the jaws for implant treatment planning. A CBCT scan was taken using a Planmeca Promax X-ray  unit. Upon reviewing the scan, multiple small nodules of high density were identified as an incidental finding during the interpretation of the patient’s scan Figure .The nodules appear to be dispersed among the layers of the face, which were clearly displayed in more than coronal and axial cuts Figure .The multiple nodules were consistent with the miliary type of OC and his past history of acne as a teenager. The palatine tonsils, in addition, revealed radiopaque masses bilaterally, but more numerous and prominent on the left side. The calcifications were also consistent with the patient history of recurrent tonsillitis as tonsillar stones (tonsilloliths) Figure . The patThe case reported in this study is that of OC with tonsilloliths, diagnosed using CBCT. OC is a benign and rare condition in which soft-tissue ossifications occur in the dermis layer of skin. The disease is categorized into primary or secondary forms. Primary forms are characterized by the lack of any history that would lead to predispose bone deposition within the layers of the skin, such as trauma or cutaneous disease, and accounts for 15% of OC cases . SecondaTheories, ranging from hamartomas to nevoid tumors, have been associated with the etiology and pathogenesis of OC . Also, iClinically, OC presents with different asymptomatic forms. It may present as single or multiple papules, nodules, or plaques or as miliary lesions . The lesUnderstanding the radiographic presentation of OC facilitates definite identification since several imaging features may be mimicked by other soft-tissue calcification conditions confounding accurate diagnosis. For example, myositis ossificans, calcinosis cutis, osteoma mucosae, and dermal fillers may have the same radiographic manifestations , 15-16. In this case report, OC is combined with tonsilloliths. The specific etiology and pathogenesis of tonsilloliths remain unknown, however, it is generally agreed that unresolved tonsillitis is the main cause . RecurreDifferential diagnosis of tonsilloliths includes mostly sialoliths, lymph node calcification, and phleboliths. CBCT images provide bilateral views of the jaw and are useful to determine and differentiate the locations of these calcifications .Correct diagnosis of the different types of soft-tissue calcifications requires sufficient knowledge from the dental professional. Clinical management of these conditions must be based on the patient’s medical history and symptomatology . VolumetIn this case report, we presented a rare case of OC, combined with tonsillolith using CBCT as a diagnostic method. The general dentist, using three-dimensional dental radiography, typically encounters soft-tissue calcifications. Based on careful examination of the patient’s scan, incidental findings are adding to the value of using CBCT as a diagnostic imaging tool and ultimately, improved patient health.