Promyelocytic leukemia is a known medical emergency and requires rapid diagnosis and expedient therapy with differentiating agents. We present an unusual case in which the diagnosis is based on a fine needle aspirate of a humeral mass. Despite lack of systemic involvement, the sarcoma responded to traditional differentiation agents. Acute promyelocytic leukemia (APL) is defined by the World Health Organization (WHO) as a subtype of acute myeloid leukemia (AML) with recurrent cytogenetic abnormalities, typically demonstrating t(q22q21) involving the fusion of the promyelocytic leukemia gene (PML) and the retinoic acid receptor‐alpha gene (RARA). Patients often present with disseminated intravascular coagulopathy (DIC) with leukocytosis or leukopenia. It is imperative that this diagnosis be histologically and cytogenetically confirmed so that differentiation therapy with all‐trans retinoic acid (ATRA) and arsenic trioxide can be promptly started. Although these patients usually have excellent clinical outcomes if treated expeditiously, unusual clinical presentations may cause delayed diagnosis and treatment. In this report, we present a patient with promyelocytic sarcoma with no peripheral blood or iliac crest bone marrow involvement by leukemia. The diagnostic challenge, treatment, and clinical course are of particular interest given the unique clinical circumstances.A 52‐year‐old woman was seen in the outpatient orthopedics clinic for evaluation of right upper extremity swelling and pain of 3‐week duration (pain score of six at its worst), which initially extended from her right shoulder down to the fingers, and resolved without treatment, albeit with residual pain at presentation. The patient reported subjective fevers that occurred at the onset of swelling and spontaneously resolved within 24 h. Physical examination of the right shoulder revealed full active and passive range of motion with mild tenderness over the greater tuberosity of the right humerus. Laboratory tests including complete blood count with differential, and serum protein electrophoresis were unremarkable except for elevated erythrocyte sedimentation rate (ESR) and C‐reactive protein (CRP). Magnetic resonance imaging (MRI) of the right shoulder demonstrated a nonspecific intramedullary mass in the greater tuberosity of the proximal humerus, measuring 3.2 × 2.1 × 2.8 cm , and RT‐PCR was negative for the PML/RARA fusion in both the peripheral blood and bone marrow specimens. These findings confirmed that the patient had promyelocytic sarcoma involving the right humerus but without the typical leukemic presentation of APL.Additional CT scans performed for staging revealed abnormally increased metabolic activity in the sternum and manubrium, acromioclavicular joints, left acromium, right humeral head with mild increased osseous density, bilateral humeral diaphysis, bilateral proximal ulnar and radial heads, bilateral distal femurs, right distal femoral metaphysis, and bilateral proximal tibias (SUVs ranging from 7.4 to 27.4). No other abnormalities were noted.The patient was started on treatment as per Lo‐Coco et al. with all‐trans retinoic acid (ATRA) and arsenic trioxide which she tolerated well without any complications Herein, we describe the case of a patient diagnosed with APL with a highly unusual presentation. Neither the peripheral blood nor the bone marrow aspirated from the iliac crest was positive for APL characteristic gene rearrangement, or had the morphologic features of APL. The aspirate from the right humeral mass, however, was positive for these APL‐defining features, thus resulting in a diagnosis of primary promyelocytic sarcoma.Acute promyelocytic leukemia rarely presents as a mass‐forming lesion without evidence of leukemic involvement, thus making this a highly unique case. Only a few cases have been reported in primary APL presents as a solitary lesion Isolated myeloid sarcoma has often been misdiagnosed as a malignant lymphoproliferative disorder, Ewing sarcoma, or other hematopoietic neoplasm, often due to inadequate immunophenotyping of the malignancy The unusual presentation also raises questions regarding treatment and prognosis of such patients. It is unclear whether the same propensity for DIC exists in patients who lack leukemic manifestation of the disease. Additionally, it would of interest to determine if there are additional genetic or epigenetic changes within the leukemic cells which allowed for such an unusual presentation. At present, it is of great importance that the patient has responded well to traditional APL regimens as assessed by imaging studies.SS: finalized the writing of the manuscript with an emphasis on the pathologic elements and integrating radiographic images into the report. NH: finalized the writing of the clinical portion of the manuscript. DLD: contributed the radiographic images, contributed to interpretation, and helped edit the manuscript to reflect radiographic findings. HK: wrote the early draft of clinical history. VG: contributed to the clinical portion of the manuscript. AE: treated the patient and was involved in final manuscript editing with Rima Koka. RK: diagnosed the patient and was involved in final manuscript editing with Ashkan Emadi.None declared.


We report a rare case of an aggressive osteoblastoma (OB) involving the calvaria and infiltrating the dura, a finding that was not previously reported in the literature. A 50-year-old man presented with a progressive mass in the left frontoparietal skull with headaches and a six-month history of sudden mass growth. Computed tomography (CT) and magnetic resonance imaging (MRI) showed a large skull lesion with areas of hemorrhage, calcification, restricted diffusion, and enhancement.  A left temporoparietal craniotomy with a complete resection of the tumor with grossly clean margins was performed. Follow-up at 60 months showed a stable clinical picture and no sign of local recurrence of the lesion on MRI. Osteoblastoma is a rare benign bone-forming tumor that accounts for less than one percent of all primary bone neoplasms  and occuBased on our literature research, only eight cases were related to calvarial aggressive osteoblastomas, when considering the frontal, parietal, temporal, and occipital bones as true calvarium . We repoA 50-year-old man presented with a progressive mass in the left frontoparietal skull with headaches and a six-month history of sudden mass growth of a previously stable left subcutaneous parietal mass present since a fall at the age of six years. Computed tomography (CT) and magnetic resonance imaging (MRI) of the brain showed a large skull lesion with areas of hemorrhage, calcification, restricted diffusion, and enhancement. Intraoperative findings showed dural infiltration through the cortical side and large vessels from the cortex feeding the infiltrated dura. The histological examination revealed an aggressive osteoblastoma (OB) with a secondary aneurysmal bone cyst.His past medical history included coronary artery disease stented the year prior, hypertension, and remote bowel surgery for a perforated viscus. Physical examination revealed a firm, non-tender, slightly mobile mass in the left parietal area. Cranial nerves were intact, there was no pronator drift, and sensation and power were normal in all limbs.Preoperative radiological evaluation included CT with and without contrast, and MRI Figure .The solitary destructive left frontoparietal bone lesion was made of intracranial (3.9 x 6.7 x 4.6 cm) and extracranial components 4.7 x 2.4 cm) destroying both the inner and outer tables. The dura directly under the mass showed a signal change and thinning. The mass was heterogeneous with loculations and septations, fluid-line, and hemorrhagic or calcified areas, with no evidence of surrounding brain edema. There was a 7 mm midline shift. A chest CT showed several lung nodules measuring up to 8 mm that were eventually deemed not related to the cranial lesion. The metastatic workup, as well as blood tumor markers, were negative. The vascularization of the tumor was assessed through an angiogram that revealed a well-vascularized lesion with shared blood supply between the middle meningeal artery and the superficial temporal artery. Branches of the middle meningeal artery were embolized the day prior to surgery . Macroscopically, the mass contained large hemorrhagic areas, calcified nodules, and bony spicules. The rest of the tumor included yellowish connective tissue on the outer aspect and adipose-like grey tissue Figure . MicroscThere was no neurological deficits postoperatively. A custom-made synthetic bone flap was inserted 56 days after the initial surgery. Follow-up at 15 months showed a stable clinical picture and no sign of local recurrence of the lesion on MRI Figure .Osteoblastoma is a rare locally aggressive cancer originating in the medulla of the bone with a high rate of local recurrence. The involvement of the calvarium happens only in four percent of osteoblastoma cases . After rAs a consequence of the rare nature of this lesion, there are no standardized management options for an aggressive osteoblastoma of the calvarium. Generally, gross total excision is adopted as the surgical treatment for aggressive osteoblastomas and could be achieved by curettage or by en bloc resection. Complete excision decreases the recurrence rate and the propensity for malignant transformation. However, in some contexts, total excision cannot be achieved due to the tumor‘s location, and adjuvant therapy may have a useful role.Aggressive osteoblastoma is a rare locally destructive bone lytic tumor, which is not included in the same spectrum of osteosarcosis. Commonly, the diagnosis cannot be made intraoperatively through frozen sections; thus, a precise histological evaluation is essential for accurate diagnosis and management. The radiological findings are variable and can be nonspecific, showing similar patterns to other tumors. We add a ninth case of aggressive osteoblastoma of the calvaria to the eight cases already reported in the literature. This is the first reported case of dural invasion.There are no standard management options after surgical resection. Wide margin resection and duraplasty should be adopted depending on dural involvement. Adjuvant therapy is reserved for non-satisfactory resections and tumors with malignant transformation to osteosarcoma. Follow-up should be continued for at least for five years due to the risk of local recurrence.  Our patient's five-year follow-up study revealed no evidence of tumor recurrence.


Low-grade myofibroblastic sarcoma (LGMS) is a malignant lesion composed of myofibroblasts. It is an uncommon tumor of unknown etiology that mainly develops in the bone or soft tissue and is most often reported in the head and neck, particularly in the tongue and oral cavity.A 2-year-old girl, previously well and with no significant medical history or family history of other diseases, presented with a 2-week painless swelling of the right orbit.Preoperative computed tomography (CT) revealed a large homogeneous enhanced mass, 21 × 13 mm in size, located on lateral wall of the right orbit with bone absorption. The mass was resected and histopathological examination revealed LGMS of the orbit.On May 2016, she underwent surgery without the additional postoperative treatment.The patient's postoperative course was uneventful, and was discharged on the 6th day after surgery. During a year follow-up period, there was no recurrence of the postoperative CT. The patient and her family were satisfied with the result of the surgery.Based on clinical characteristics and postoperative CT, we considered the mass may be a benign tumor. We completely resected along the capsule without an extensive surgical margin. However, postoperative histopathology diagnose LGMS, which shows a strong potential for local recurrence and vascular invasion. So we should close observation of the patient's symptoms and sign. If the tumor has invaded adjacent tissues, we will use adjuvant chemotherapy or radiotherapy. LGMS consists mainly of myofibroblasts, which are spindle-shaped mesenchymal cells with ultrastructural features of both fibroblasts and smooth muscle cells. Some myofibroblastic tumors have relatively benign or only low-grade malignant characteristics. LGMS has been most frequently reported in adult men, and has been found in a wide variety of tissues, including skin, breast, vulva, salivary glands, parapharyngeal space, jaw, larynx, nasal cavity/paranasal sinuses, soft tissue of the cheek, piriform fossa, soft palate, and tongue.–18 Reports of LGMS of the orbit are extremely rare. Here, the authors report a case of a 2-year-old girl with LGMS of the orbit and discuss its clinical manifestations, pathological characteristics, diagnosis, therapy, and prognosis. The previous literature is reviewed.Myofibroblastic sarcoma was reported as a distinct entity in 1998 by Mentzel.2This study was approved by the Ethics Committee of Shengjing Hospital of China Medical University. Written informed consent was obtained from the patient's parent. A 2-year-old girl, previously well and with no significant medical history or family history of other diseases, presented with a 2-week painless swelling of the right orbit Fig. A and B. The patient underwent open surgery under general anesthesia for excision of the mass, which was exposed via a right lower eyelid approach. The mass had an intact capsule measuring 2.5 × 2.5 × 1.5 cm and was completely resected along the capsule. Macroscopically, the tumor was round, firm, smooth, and well circumscribed, measured approximately 2.0 × 1.5 × 1.5 cm, and had a yellowish-gray and homogeneous surface on transection Fig. A–D.Histopathologic examination confirmed the diagnosis of LGMS. Microscopic examination showed spindle-shaped tumor cells that were arranged in a fascicular or storiform pattern infiltrating the adjacent bone trabecula. The cells were demonstrated nuclear atypia with mitotic figures. The cytoplasm was acidic and the stroma consisted of a small number of collagen fibers. On immunohistochemical staining, the spindle-shaped cells were strongly and diffusely positive for smooth muscle actin (SMA) and vimentin Fig. C and D. The postoperative course was uneventful and the patient did not receive adjunctive therapy. The patient was discharged on the 14th day after surgery. No additional treatment was necessary, and there was no recurrence or metastasis of the postoperative CT during a year follow-up period Figs.  and 6.3 LGMS is a low-grade mesenchymal malignancy composed of myofibroblasts that can occur in submucosal and subcutaneous tissues, deep soft tissues, and intraosseous areas. Myofibroblasts are modified fibroblasts that are morphologically and functionally similar to both fibroblasts and smooth muscle cells. They were first reported in granulation tissue and can also arise in the stroma of normal, inflammatory, neoplastic, or reactive soft tissue lesions.,22 LGMS shows a strong potential for local recurrence, while vascular invasion and malignant transformation occur only occasionally. In some cases, LGMS has an incomplete capsule and may infiltrate adjacent fibrous tissue, fat, or skeletal muscle.The World Health Organization classification of soft-tissue tumors describes LGMS as an atypical myofibroblastic proliferation with fibromatosis-like features that most commonly develop in the head and neck. The mean age of affected patients is 40 years, ranging from 4 to 85 years, and a preponderance of adult men is reported. The tumors generally present as a painless swelling or a slow-growing mass with a relatively indolent course. Some patients may present with pyrexia, chills, leukocytosis, or meningeal irritation.While LGMS is most commonly found in the head and neck, particularly in the tongue and oral cavity, it has also been reported in almost every other organ. Therefore, new molecular imaging may be an effective method for preoperative diagnosis, therapy as well as prognosis in malignant tumors, especially occurred in the head and neck regions. Of molecular imaging methodologies, 3 main techniques are intriguing. The first one is nuclear medical imaging, in which radioactive molecular probe is utilized and its distribution and kinetics are measured by positron emission tomography (PET) and single-photon emission computed tomography. Probes for nuclear molecular imaging were developed to image intact function difference between wild-type and the transduced type F98 gliomas in cat brain. 18F-labeled amino acids for PET/CT imaging were utilized to show elevated amino acid metabolism in gliomas, neuroendocrine tumors, prostate cancer, and breast cancer.–29 A number of amino acid PET radiotracers were helpful in pretreatment evaluation of pediatric low-grade gliomas. The second one is optical molecular imaging, in which a fluorescent probe is used to provide real-time imaging of distribution and kinetics. Although high spatial resolution could be obtained inexpensively, the imaging region is limited to the body surface. The third one is photoacoustic imaging, which could detect highly sensitive and high-resolution photoacoustic signals in deep tissues. Thus, we speculated that new molecular imaging techniques could be helpful for preoperative diagnosis, therapy, and prognosis estimate in LGMS. Unfortunately, due to the lack of the new molecular imaging system, this patient was not performed these specific examinations before surgery. Because cytogenetic and molecular genetic alterations of LGMS are presently obscure, in vivo molecular imaging could be tested in this disease in the future.Although preoperative CT or magnetic resonance imaging is essential for diagnosis, LGMS is hardly diagnosed definitely by them. In vivo molecular imaging is an innovative and cornerstone method for clinical diagnosis for tumors. The new method collects information at the molecular and cellular levels in humans. Histologically, LGMS is mainly composed of slender spindle cells showing low mitotic activity and variable nuclear pleomorphism. The nucleus has been described as fusiform, slender, and undulate. The tumor cells are arranged in sheet-like interlacing fascicles or storiform patterns, and have eosinophilic cytoplasm with sparse inflammatory lymphocytic and plasmacytic infiltration. The interstitial tissue may consist of hyalinized collagen fibers. Immunohistochemical staining may be positive, to some degree, for SMA, muscle specific antigen, fibronectin, desmin, calponin, and vimentin. CD34 and CD99 are also positive in a small fraction, while anaplastic lymphoma kinase (ALK), S-100, and epithelial markers, including cytokeratin and epithelial membrane antigen, and laminin are generally negative.,6,35A definitive diagnosis of LGMS is made mainly on the basis of histopathological findings and immunohistochemical analyses. Because LGMS may have a tendency for invasive growth and local recurrence, an extensive surgical margin should be recommended, although in our case, the mass was completely encapsulated. Complete resection of the primary lesion and local recurrent lesions is essential, but the optimal extent of resection has yet to be determined. Some authors have recommended adjuvant chemotherapy and radiotherapy, particularly if the tumor has invaded adjacent tissues or the lymphatic or hematological metastasis is evidenced. However, other reports have indicated that myofibrosarcoma is poorly responsive to radiation therapy, and that the cure rate with chemotherapy is uncertain. Thus, the best method of treatment requires further prospective investigation. We did not recommend postoperative chemo- or radiotherapy for the present patient because of concerns regarding side effects and preservation of her eyesight.Surgery is the primary treatment modality for LGMS. Fibrosarcomas are composed of malignant spindle cells showing fibroblastic differentiation, which are distinguished from myofibroblasts by the absence of immunohistochemical evidence of fibronectin, SMA, and calponin. Leiomyosarcoma is characterized by alternate fascicles of cells with marked cytological pleomorphism that show immunoreactivity for desmin, h-caldesmon, and occasionally keratin, without evidence of immunoreactivity for fibronectin. Nodular fasciitis is less cellular and uniform than LGMS, and has a heterogeneous appearance with cellular, myxoid, and fibrous areas. Fibromatosis may display a prominent nodularity, and tends to infiltrate adjacent tissues. The tumor cells are lacking of atypia and mitotic figures, and are positive only for vimentin. Finally, LGMS is most easily confused with IMT. IMT is mainly composed of myofibroblastic spindle cells mixed with prominently lympho- and plasmacytic infiltration, and is positive for ALK or cytokeratin. while LGMS has a more uniform histological pattern, with a greater degree of nuclear atypia and more mitotic figures, and it is negative for ALK and cytokeratin at immunohistochemistry.The differential diagnosis of LGMS includes a variety of malignant or benign lesions, including fibrosarcoma, leiomyosarcoma, inflammatory myofibroblastic tumor (IMT), nodular fasciitis, and fibromatosis.4In the present report, we describe a rare case of LGMS in the wall of the right orbit in a 2-year-old girl. The tumor was completely encapsulated and followed a relatively indolent course. Complete resection was performed. The prognosis was good, and at a year after surgery the patient remained recurrence-free. LGMS presents many diagnostic challenges, particularly in the differentiation of a low-grade malignant process from an intermediate-grade malignant transformation. In vivo molecular imaging may be an effective method for diagnosis and postoperational follow-up for LGMS, and should be further evaluated.


Pheochromocytoma is a tumor which originates from chromaffin cells of the adrenal medulla or the sympathetic ganglia. This tumor secrets a high amount of catecholamine and metabolites, causing hypertension crisis with headache, tachycardia, sweating and flushing (classic triad of pheochromocytoma). However, in some cases the disease may cause atypical symptoms or may be asymptomatic. The presented patient is a 34-year-old man who referred to our clinic with left flank pain. He had a history of falling from height. In the primary physical examination, a large mass in the abdominal left upper quadrant was palpated. After diagnostic evaluation malignant pheochromocytoma was detected. The patient was discharged on the fourth day after surgery. Malignant pheochromocytoma can presented with atypical symptoms or can be asymptomatic. Pheochromocytoma is a tumor in medulla region of adrenal gland which cause hypertension, headache and tachycardia. The presented patient is a 34-year-old man who referred to our clinic with left flank pain. He had a history of falling from height. In the primary physical examination, a large mass in the abdominal left upper quadrant was palpated. After diagnostic evaluation malignant pheochromocytoma was detected. The patient was discharged on the fourth day after surgery. Malignant pheochromocytoma can presented with atypical symptoms or can be asymptomatic.Pheochromocytoma is a tumor which originates from chromaffin cells of the adrenal medulla or the sympathetic ganglia. The annual incidence of the disease is 1.6 to 2.1 per million. Around 90% of the cases are sporadic and occur among women. Other cases are young men with a family history and the multiple endocrine neoplasia (MEN) syndrome  scan, there was a 122×107 mm heterodense and lobulated soft tissue mass with central necrosis in the left supra renal region  and anotLiver, spleen, pancreas and other viscera inside the abdomen and pelvis were normal. Metaiodobenzylguanidine (MIBG) scan showed multiple absorptive lesions in the liver and left lung. Echocardiography was performed which was normal. The patient was diagnosed with malignant pheochromocytoma and initially was treated with phenoxybenzamine (60 mg/day) and atenolol for 3 weeks and dextrose saline 3 days prior to surgery. With a subcostal incision, a radical surgery including left adrenal mass removal, left nephrectomy and para-aortic lymphadenectomy was performed . Sodium Pheochromocytoma is a tumor which originates from adrenal chromaffin cells or extra-adrenal (paraganglioma). Its classic symptoms include headache, sweating, tachycardia and paroxysmal hypertension. These symptoms are due to the production and secretion of catecholamine, frequently caused by the tumor. The patient had no objection of any typical cardiovascular symptom. However, following a mild trauma due to falling from low height, he suffered from flank pain and reported a history of headache, sweating, nausea and weight loss in the past months prior to his referral.In one study it was demonstrated that the classic symptoms of pheochromocytoma are more common among the benign type while backache and abdominal pain are more common in the malignant type . The besLaboratory tests including urine and plasma catecholamine measurement, urinary metanephrine and vanillylmandelic acid (VMA) have a sensitivity of about 76%. Measurement of plasma free metanephrine is the latest available test. Since secretion of catecholamine is periodic and proximal, a single measurement may lead to a false negative. Thus, sensitivity increases with repeated tests (two or three times), particularly following incidence of symptoms . Other vCytological and histological appearances of benign and malignant pheochromocytoma are often the same. Malignancy is only confirmed by invasion or metastasis ,6. In ouOther measures include fluids replacement with sugar-salt solutions to prevent orthostatic and postoperative hypotension and hypoglycemia after surgery . Any incIn this disease, surgery and complete tumor resection is the basic and primary treatment. After surgery, the patient should be monitored for 24 hours in ICU. Two major complications after surgery include hypotension and hypoglycemia. Sudden drop in blood pressure caused by the abrupt fall in circulatory catecholamines and hypoglycemia are related to rebound hyperinsulinemia due to recovery of insulin secretion after tumor resection  have been completely observed by the authors.None.


We present the case of a young female on oral contraceptives (OCs) who was diagnosed with focal nodular hyperplasia (FNH) and remained on oral contraceptives. Months later, the patient presented with acute abdominal pain and intratumoral hemorrhage in the liver. The patient was taken to the operating room (OR) and was diagnosed with a ruptured hepatic adenoma (HA). We review the key diagnostic features of FNH and HA, the different management guidelines including use of OCs, and potential surgical indications. HA compared to FNH has a significantly higher rate of sequelae despite being a benign lesion, thus providers must accurately distinguish between the two diagnoses to prevent potential morbidity and mortality. After hepatic hemangioma, focal nodular hyperplasia (FNH) and hepatic adenoma (HA) are the second and third most common benign liver neoplasms, respectively. Both tumors are typically asymptomatic and are usually discovered incidentally on imaging. HA is strongly associated with exposure to estrogens, particularly estrogen-containing oral contraceptives (OCs) . OverallA 31-year-old Caucasian female presented to the emergency department with acute right upper quadrant (RUQ) abdominal pain over the past day. The patient described her pain as constant, sharp, and burning, with radiation to the back. The patient also complained of nausea and non-bloody, non-bilious emesis. Her past medical history was significant for hypertension and a liver mass consistent with FNH that was incidentally noted on abdominal computed tomography (CT) over a year ago  to review the liver mass findings and potentially receive outpatient magnetic resonance imaging (MRI), the patient did not follow up. The patient’s relevant medications were OC use for the past six years. On physical examination, there was hepatomegaly and significant tenderness to palpation over the RUQ. The patient did not exhibit rebound tenderness, guarding or Murphy's sign.Initial work-up included a negative pregnancy test, elevated liver enzymes with an Alkaline phosphatase level of 116 IU/L, Aspartate Aminotransferase (AST) of 160 IU/L, Alanine Aminotransferase (ALT) of 281 IU/L, and total bilirubin of 0.5 mg/dL. White blood cell count (WBC) was slightly elevated at 12,000 per microliter and her hemoglobin level was 11.5 g/dL. The international normalized ratio (INR) was 1.1 and Creatinine was 0.77 mg/dL. CT scan of the abdomen with contrast demonstrated the same large heterogeneous mass in the right lobe of the liver  or FNH.The patient underwent a laparoscopic-assisted extended right hepatectomy including parts of segment 4 and 1 . Currently, it is the second most common cause of benign hepatic tumors in adults behind hepatic hemangiomas . AlthougHistologically, FNH is described as a focal form of cirrhosis with a central fibrous scar, ductular proliferation, and malformed vessels. The central fibrous scar may manifest on CT scan or MRI as a central stellate scar, which is pathognomonic for FNH. Other imaging characteristics typically include a mass less than 5 cm in size . Though FNH is typically asymptomatic; the lesion is usually found incidentally, and only 12-13% of patients reporting abnormal serum liver tests. Rare symptoms include palpable abdominal mass (2-4%), hepatomegaly (<1%), and fever (<1%) .Behind hepatic hemangioma and FNH, HA is the third most common cause of benign liver neoplasms and is found almost exclusively in women between the second to fifth decade of life. Since the 1970s, with the advent of OCs, the incidence of HA in young women has increased .HAs are reported to be solitary lesions (70-80% of cases), which lack an obvious fibrous capsule on imaging. Histologically, these neoplasms are characterized by the presence of liver cell plates in the absence of bile ducts and fibrosis . AberranLike those with FNH, a majority of patients with HA are often asymptomatic with up to 7% of cases reporting abnormal serum liver tests. Thirty to 40% of patients report abdominal discomfort and 2-4% have a palpable abdominal mass . These pVarious epidemiologic, histologic, diagnostic, and clinical management differences exist between FNA and HA, and these differences are highlighted in Figure In our case, the patient was diagnosed with an FNH based on CT imaging. An MRI was recommended for followup, but the patient did not follow up and did not receive additional imaging. As discussed, atypical lesions may be difficult to diagnose on CT and thus require MRI. A diagnosis of HA could have led to earlier termination of her OCs. Although unlikely to have prevented surgery, due to the large size of the tumor, appropriate followup and imaging may have led to the reduction in the size of the liver resection and higher likelihood of complete laparoscopic resection.Although HA and FNH are common hepatic lesions with numerous similaries, clinicians should be aware of the diagnostic differences, preferred imaging modalities, and clinical management differences. Clinicians should have discussions with their patients with regard to the use of OCs, the necessity of close outpatient followup, choice of imaging modalities, and the potential need for surgical consultation.


Lipoma is the most common benign tumour of the human body, being intraosseous involvement very rare. Just 1 to 4% of all cases of lipoma are located in the oral cavity, only 0.1% being intraosseous. The jaw is its most uncommon bone location. Etiology of intraosseous lipoma (IOL) is unknown, although several theories have been proposed. Usually asymptomatic, the symptoms, when present, will depend on its location and size. Its origin may be intraosseous or juxtacortical. A biopsy is essential for diagnosis, and definitive treatment involves resection or curettage of the lesion. The aim of this paper is to present a new case of intramedullary intraosseous lipoma of the mandible with involvement of the left mandibular ramus and condylar neck.A case of intramedullary intraosseous lipoma (IOL) on the left mandibular ramus and condyle is presented. No history of trauma in temporomandibular joint existed. The radiology showed a radiolucent multi-lobulated lesion with values of attenuation in the range of fat. Curettage is performed and the histopathology showed a conglomerate of adipocytes without trabeculae, calcifications or atypia.According to the bibliography 24 cases of mandibular IOL have been described. This is the second reported case of condylar involvement and the first with cortical expansion.Lipoma intraosseous is a very rare benign bone neoplasm. Histology is required for the differential diagnosis from other radiolucent lesions. The IOL treatment is the curettage with a good prognosis, although malignant transformation to liposarcoma has been reported in other locations. It is a disease with a difficult differential diagnosis, therefore the publication of new cases is important. Key words:Intraosseous lipoma, lipoma, jaw tumour, condylar tumour. Lipoma is a circumscribed, slowly growing benign mesenchymal tumour, formed by a conglomerate of mature adipocytes without cell atypia. It can be found in multiple locations due to the wide distribution of fat tissue throughout the body. Subcutaneous lipoma is the most frequent clinical presentation, but lipoma may be also found at intramuscular, retroperitoneal and intraosseous levels . Oral caThe first case of mandibular intraosseous lipoma (IOL) was reported by Oringer in 1948 as a radiolucent lesion under a second molar; when chewing or exerting pressure in the molar region pain was elicited ,7. The mA 50 year-old female was referred to our service because of a radiolucent image on the left mandibular ramus and condyle discovered in a radiographic examination. No history of temporomandibular joint trauma or pathology was present. An orthopantomography showed a radiolucent, multi-lobed, well circumscribed lesion, in the left mandibular ramus and condyle Fig. . A compuMandibular IOL is very rare, with only twenty-three cases reported in the literature . Only onIn 1969, Hart studied the location of intraosseous lipoma and its relationship to the bone, establishing four categories: lipoma from the surrounding soft tissue, intraosseous lipoma of the intramedullary cavity, periosteal lipoma which may deform the bone by pressure or periosteal reaction, and the liposarcoma, a malignant tumour with local destruction and possible distant metastasis . In 2001Etiology of IOL remains unclear; dental trauma, disruption of the post-extraction healing process, retention of radicular remains , medullaThe intramedullary mandibular IOL shows up on x-rays as a well-circumscribed radiolucent uni- or multi-locular lesion, with possible central calcification or ossification and a partial or complete sclerotic border, which may or may not be associated with cortical expansion. A CT scan shows a homogeneous density equal to the density of fat, although heterogeneity may also appear when myxoid degeneration or calcification develops. Marginal sclerosis and cortical irregularity can also be seen, although re-sorption is a rarity. In our case, a lytic image producing a thinning of cortical bone and a focal disruption of the vestibular cortex in the mandiblar ramus was seen. On MRI a similar homogeneous signal intensity with fat density was seen in both T1 and T2. No cases of multiple bone lesions have been reported .The histopathology is characterized by presence of mature adipocytes without atypia and hematopoietic tissue, which may be encapsulated. Milgram established three stages based on the viability of the fatty tissue and the presence of calcification or bone trabeculae: stage 1 presents absence of them, stage 2 presents partial necrosis and calcification, with viable adipose tissue remaining, and stage 3 presents complete necrosis and tissue involution, with dystrophic calcification, and a greater predisposition to malignancy ,15. Our The differential diagnosis must be performed with other radiolucent bone images; such as the simple cyst, post-traumatic cyst, aneurysmal bone cyst, giant cell granuloma, ameloblastoma, osteoblastoma, arteriovenous malformations, hemangiomas, infarcted bone, chondrosarcoma or liposarcoma -4.The treatment consists of curettage with or without bone graft or filling, which in our case was not necessary. Other techniques have been used such as phenolization, without much success with regard to the surgery. Only four cases of malignancy localized in the tibia, fibula and femur have been reported, but no case of mandibular IOL malignant transformation or recurrence . In conc


Gynaecomastia is the most common benign breast disorder in men. Among the various causes, testicular malignancies are an uncommon, life-threatening condition requiring prompt diagnosis and treatment. The case of a 28-year-old man is discussed, who presented with a 6-month history of painful bilateral gynaecomastia with no abnormality on clinical or biochemical examination. The patient's symptoms spontaneously resolved within 4 weeks. He then represented 10 years later with similar symptoms and an associated secondary hypogonadism. Ultrasound imaging revealed a clinically occult, hypoechoic mass in the left testis (Leydig cell tumour on histology). Clinical and hormonal findings normalized following surgical excision. This report underlines the importance in clinical practice of ultrasonographic evaluation of the testis, in all patients with gynaecomastia, despite unremarkable findings on physical examination.


Intra-articular fibroma of tendon sheath is a rare disease. To our knowledge, less than 20 cases have been reported in the literature, and none of them was a Chinese patient. In this case report, we present a Chinese patient with intra-articular fibroma of tendon sheath of the knee joint which was excised arthroscopically. We also summarized the clinical presentation, diagnosis, and subsequent management of intra-articular fibroma of tendon sheath. Fibroma of tendon sheath (FTS) is a rare dense fibrous benign tumor that majorities are found over the tendons or tendon sheaths of limbs, with predilection in order starting with fingers, hands, wrists, and other parts.A 36-year-old male patient had complained a painless mass over the suprapatellar region of left knee for 2 months. He did not complain any systemic upset, and he did not recall any trauma history. Physical examination showed a palpable mass over a suprapatellar region of left knee, which was mobile and firm. The range of motion of the left knee was from 0 to 110 degrees. The joint line was not tender. All special tests including McMurray test, Lachman test, and grinding test of patella were negative.X-ray examination of the knee showed no bony abnormality. Preoperative magnetic resonance imaging (MRI) of the left knee showed a suprapatellar mass measured 3 cm × 2 cm, hypointense in T1-weighted image  but hypArthroscopic examination of left knee was performed under spinal anesthesia. The tumor was located at the suprapatellar region as shown in MRI. The isolated circumferential mass was grayish, and there was a stalk attaching to the superior pole of the patella . The tuFTS is most commonly found in adults, peaking at 30 to 50 years old and males are commonly affected.X-rays are usually normal, except the tumor is very large compressing the surrounding bony tissue causing erosion.In conclusion, intra-articular FTS is a rare disease, but it should be considered as one of the differential diagnosis of a soft tissue tumor arising from the knee joint. MRI is necessary for diagnosis of intra-articular FTS. Simple excision either open approach or arthroscopy can cure the disease. Regular follow-up with interval imaging is recommended as recurrence rate is relative high.


Small bowel obstruction (SBO) is a common illness encountered by general surgeons. However, obstruction caused by diaphragm disease induced by non-steroidal anti-inflammatory drug (NSAID) is exceedingly rare. The diagnosis is challenging as the signs and symptoms are neither sensitive nor specific. We report the case of a 59-year-old male who presented with SBO secondary to this uncommon condition. We hope to raise awareness of this unusual entity. Small bowel obstruction (SBO) is a common condition treated by general surgeons. It can be precipitated by a myriad of factors, broadly divided into extrinsic, intramural, or intraluminal conditions. SBO secondary to diaphragm disease induced by non-steroidal anti-inflammatory drugs (NSAID) is exceptionally rare. We discuss the case of a 59-year-old male who presented with this uncommon entity.A 59-year-old male presented to the emergency department with a three-week history of worsening colicky central and lower abdominal pain. The pain was exacerbated by food. His bowels had been opening regularly in small amounts with the aid of laxatives. He had a 2 kg weight loss over the last few months. He had osteoarthritis of the knee, for which he had started using meloxicam 15mg daily for the last nine months. He was otherwise generally fit with no other medical or surgical history.On examination, he was afebrile and hemodynamically stable. His abdomen was tender in the right lower quadrant and peri-umbilical region; there were no signs of peritonism.9/L, haemoglobin 128 g/L, liver functions and renal functions were all within normal limits.His blood tests showed C-reactive protein 9.6, white cell count 9.6 x 10He proceeded to have a computed tomography (CT) scan of the abdomen. This showed a partial SBO with dilatation of the mid to distal small bowel and faecalisation of intraluminal material in the distal segments. There was a transition point in the right iliac fossa with a partial collapse of the distal small bowel Figure .He had a trial of 24 hours of conservative management, which included clear fluid diet, analgesia, and anti-emetics. His symptoms worsened so he proceeded to have a diagnostic laparoscopy on day two of admission. At laparoscopy, he had a copious amount of serous free fluid in the abdomen and dilated small bowel with evidence of congestion. However, no clear cause of bowel obstruction was identified. The decision was made to convert to mini-laparotomy to deliver the small bowel and palpate for any intrinsic lesions. Two concentric strictures were felt in the distal jejunum and these were observed to be the cause of obstruction. The strictured small bowel was resected and an end to end hand-sewn anastomosis was performed. The mesenteric defect was closed and his abdominal fascia and skin were closed. He had a non-complicated recovery in the ward. His diet was progressively upgraded and his bowels were opened on day three post operation. He was discharged from hospital on day four after tolerating a full diet. A follow-up phone call three weeks post operation confirmed that he had made a full recovery with no further complaints.Macroscopic examination of the bowel specimen revealed a narrowed band on the serosal surface of the bowel and the underlying bowel wall felt thickened. Microscopically, there were occasional foci of mucosal ulceration which appeared to be located at the tip of folds within the small intestine. Some of these foci of ulceration were associated with granulation tissue within the lamina propria. Foci of ulceration were associated with focal crypt dropout, focal cryptitis, and crypt abscess formation. The adjacent epithelium focally showed reactive appearing atypia and occasional intraepithelial neutrophils. Immediately deep to some of these areas of ulceration, there appeared to be increased fibrosis of the submucosa. Whilst no classical diaphragms were identified macroscopically, the microscopic features were consistent with NSAID-induced diaphragm disease Figure .SBO is a common problem that has many different causes. In the context of this case, stricturing small bowel disease in a virgin abdomen can be due to a congenital band, inflammatory bowel disease, small bowel malignancy such as lymphoma, enteropathy secondary to ischemia, or in this case, diaphragm disease.First formally described by pathologist Lang in 1988 . The patDiagnosis based on history and clinical findings alone is difficult, as the symptoms and signs are usually non-specific. This is demonstrated by our patient, who presented with non-specific intermittent abdominal pain and weight loss. Imaging by CT also has limited value in confirming this diagnosis, as diaphragms can appear as exaggerated plica ciculares and they are either not seen or easily interpreted on CT . CapsuleEven at laparoscopy, findings often do not point to this specific diagnosis. The bland serosal appearance of the diaphragm often escapes the watchful eye of the surgeon. Usually, the diseased segment of the small bowel is only found by meticulous palpation of the entire length of the bowel during laparotomy.Management of SBO due to diaphragm disease involves surgical treatment and cessation of NSAID. At surgery, both stricturoplasty and resection have been reported with success. The general preference is resection because the diagnosis is often not clear at surgery . SurgicaSince SBO caused by diaphragm disease is rare, its diagnosis requires a high index of suspicion. This case highlights the importance of thorough history taking and meticulous attention to details during surgery in order to bring about a favourable outcome to this rare disease.


He underwent a high-dose methotrexate-based chemotherapy and a local irradiation therapy (40Gy). He has been alive for more than two years since the symptom onset, and without any evidence of recurrence. This case suggests that malignant lymphoma, as an infiltrating and rapidly progressive tumor, may be accompanied by syrinx.A 79-year-old man presented with primary intramedullary malignant lymphoma with a presyrinx state in the cervical cord manifesting as left hemiparesis and hemidysesthesia. The magnetic resonance imaging (MRI) scan showed an intramedullary mass in the cervical spinal cord at the level of C1 and T2-weighted image prolongation from the medulla to the level of C5. According to the progression of hemiparesis, he underwent an emergency removal of the tumor under general anesthesia. The tumor was totally removed, and the peritumoral signal abnormality was not present in the postoperative MRI. Histological examination revealed diffuse large B cell lymphoma. While brain MRI, bone marrow puncture, and  A presyrinx state may develop from a nontraumatic obstruction of the cerebrospinal fluid (CSF) pathways in the spine . While tA 79-year-old man presented with a six-month history of numbness in his left upper limb. Left motor weakness developed suddenly, and the patient was admitted to our hospital. Neurological examination on admission revealed left hemiparesis with 2/5 in his upper limb and 3/5 in his lower limb and hypoesthesia in his left side from the neck to the foot. The deep tendon reflexes were increased in his left upper limb. Magnetic resonance imaging (MRI) showed an intramedullary tumor that was homogeneously enhanced with gadolinium at the C1 level in the spinal cord  of the whole body showed no abnormalities (Figure Postoperatively, his motor function recovered to the level of 3/5 in his upper limb and 4/5 in his lower limb. Histological examination of the tumor revealed a diffuse proliferation of large atypical lymphocytes with B cell markers Figure . Based o  He has been alive for more than two years since the symptom onset, and without any evidence of recurrence.Written informed consent was obtained from the patient and his next of kin prior to any procedures.Transudation from pathological tumor vessels, secretion by tumor cells, and obstruction of the CSF flow by an intramedullary tumor might be significant factors for the development of syringomyelia. Displacing tumors are more likely to cause syrinxes than infiltrating tumors . NakamizA presyrinx state has been defined as a spinal cord parenchymal T2 prolongation that is reversible after restoration of patency of the CSF pathways . It may The most important steps in the approach to spinal cord lymphoma are biopsy and histological diagnosis followed by chemotherapy . MTX wasThis case suggests that malignant lymphoma, as an infiltrating and rapidly progressive tumor, can be accompanied by syrinx. With this in mind, intraoperative rapid pathologic diagnosis before resection should be considered for intramedullary spinal tumors with syrinx.


Dear Editor,3, as determined by planimetry. The CCT images allowed the diagnosisof degenerative caseous calcification of the mitral annulus.A 62-year-old patient with chronic kidney disease, who was undergoing treatment withintermittent dialysis, was admitted to the hospital for investigation of a complaint ofprogressively worsening dyspnea, despite the optimization of the dialysis. To elucidatethe case, ancillary tests were ordered, such tests including echocardiography. Theechocardiography showed an expansive formation in the mitral valve, and cardiac computedtomography (CCT) was performed in order to better evaluate that finding . The CCT-5. Caseous calcification of the mitral annulus is achronic degenerative process that usually involves the posterior mitralannulus. It is mostprevalent in elderly females and inpatients with chronic kidney disease who are on hemodialysis-10. It is a raredisease, accounting for only 0.5-1.0% of all calcifications of the mitral annulus.Although rare, it is one of the major differential diagnoses of cardiac tumors, thrombi,vegetations, and abscesses.Improving the use of imaging methods in the evaluation of cardiovascular diseases hasbeen the objective of a number of recent studies in the radiology literature ofBrazil. The prognosis of caseous degeneration of the mitral annulus isgood, especially in patients who are asymptomatic, although some patients develop severesymptomatic valvular dysfunction; in the latter group of patients, the prognosis is poorand surgery should be considered,12.In most cases are asymptomatic patients, and the diagnosis is established by examinationof cardiac imaging performed for other purposes. The symptoms, when present, correspondto palpitations, dyspnea, and syncope. The heterogeneity of the content of the mass wasconfirmed by the variation in its density, which can range from negative Hounsfieldunits, suggesting fatty degeneration, to elevated Hounsfield units, suggesting a highprotein content and structural calcification. The central hypointensity was secondary to liquefaction ofthe calcium that fills the center of mass,13,15.On the CCT scans, we noted a hyperintense crescent-shaped mass or a well-definedoval-shaped mass with peripheral calcification, usually along the posterior mitralannulus, which was not enhanced after contrast administrationIn this context of our findings in the case presented here, we can conclude that CCThelps confirm the diagnosis, allows the degree of mitral valve stenosis to be evaluated,and offers measures to improve treatment strategies, especially those involvingtranscatheter or percutaneous transapical mitral valve implantation. Therefore, CCT isconsidered an excellent tool for the diagnosis of caseous degeneration of the mitralannulus.


Ectopic parathyroid adenomas can occur in numerous anatomic locations. While ectopic parathyroid adenomas can rarely occur in the pharyngeal region, this has not previously been described in the soft palate.We report the first case of ectopic parathyroid adenoma within the soft palate. A 59 year old woman presented with hyperparathyroidism. She remained persistently hyperparathyroid after initial parathyroidectomy. Repeat exploration for a lesion suspicious on PET-CT for an ectopic parathyroid adenoma in the parapharyngeal region was unsuccessful in treating the hyperparathyroidism. An ectopic adenoma in the soft palate was eventually discovered. Removal through a transoral approach was successful in treating the hyperparathyroidism.Ectopic parathyroid adenomas can occur in various anatomical locations that may be missed even with the use of the various imaging modalities. The soft palate should be added to the list of possible ectopic locations high in the neck. The existence of ectopic parathyroid tissue remains a significant hurdle in the surgical management of hyperparathyroidism. Embryological development and migratory descent to the lower neck predisposes the parathyroid glands to ectopic locations. Ectopic adenomas have been reported to account for anywhere from 4 to 16 % of patients with hyperparathyroidism and are thought to be the cause of a significant portion of failed primary surgery for hyperparathyroidism .Ectopic parathyroid glands have been classically described as occurring in numerous anatomic locations anywhere from the angle of the mandible to the mediastinum –6. Most A 59 year old woman with a 7 year history of recurrent nephrolithiasis and mild hypertension presented with a complaint of weakness and fatigue with associated bone and joint pain. Further history revealed that she had had five previous episodes of urinary calculi requiring surgery or lithotripsy. She was also discovered to have had a first degree atrioventricular block and was currently awaiting pacemaker insertion. There was no family history of thyroid/parathyroid disorders or history of previous radiation. Physical examination was unremarkable. Laboratory investigations revealed hypercalcemia (2.89 mmol/L), hypophosphatemia (0.78 mmol/L) and elevations in parathyroid hormone (PTH) (15.2 pmol/L). Bone mineral density studies revealed cortical bone osteoporosis.99mTc-sestamibi scan did not reveal any abnormality. An exploratory parathyroidectomy was then arranged and the right and left superior parathyroid glands were removed, along with a localized biopsy of the left inferior parathyroid gland. The right upper gland was 1 cm in diameter and was thought to be an adenoma. No inferior right parathyroid gland was identifiable. She subsequently remained hyperparathyroid and underwent re-exploration and removal of the previously biopsied left inferior gland. Again, a right inferior gland was not identified and a right thyroid lobectomy to locate a possible intra-thyroid adenoma was performed. The final pathological analysis of the removed thyroid lobe did not identify any parathyroid tissue within the thyroid parenchyma, but there was normal parathyroid tissue in the paratracheal fat removed at the time. Parathyroid hormone levels continued to remain persistently elevated. Although subsequent MRI and CT scans remained negative, PET-CT scanning showed a 3 cm region of uptake in the right parapharyngeal region behind the thyroid cartilage at the level of the arytenoids. Selective venous sampling showed increased PTH levels in this area.A A third parathyroid exploration was undertaken for elective resection of the suspicious area shown on PET-CT. Frozen section pathology results suggested the presence of inflammatory tissue secondary to a suture granuloma from the previous neck exploration. Intraoperative PTH levels continued to remain elevated. An exhaustive search of the neck was then undertaken, including skeletonization of the carotid artery to the level of the bifurcation, mobilization of the jugular vein, identification of the course of the vagus nerve through the neck, dissection down to the cervical fascia and prevertebral fascia and mobilization of the pharynx and esophagus from the prevertebral fascia. At this point, it was thought that the lesion might be in the upper neck. A secondary incision in the upper neck was considered, but the decision was made to defer further exploration.Post-operatively, the patient continued to have elevated serum calcium and PTH and repeat venous sampling study was performed, again confirming positive uptake in the proximal right internal jugular vein. A repeat CT scan of the head and neck Fig.  was obtaThe patient underwent a successful transoral surgical removal of the lesion. Pathology confirmed the presence of a parathyroid adenoma. As expected, the patient became hypocalcemic postoperatively.The occurrence of ectopic parathyroid adenomas can typically be explained by their embryological development , 12–14. We are aware of only a few reports describing ectopic parathyroid adenomas in very superior locations. In a series of 288 patients with persistent hyperparathyroidism, Jaskowiak et al. described one patient with a lesion in the wall of the nasopharynx near the nasal septum and one patient with a lesion high in the vagus nerve at the level of the C1-C2 vertebrae . A cerviThere are sparse reports in the literature of utilizing a transoral approach to resect ectopic parathyroid adenomas. The standard surgical approach for re-exploration following persistent hyperparathyroidism is transcervical , 16. A hReoperation for persistent hyperparathyroidism can be challenging. This case demonstrates some of the challenges of surgical management of persistent hyperparathyroidism. Normal anatomy and tissue planes are often disrupted and sometimes difficult to recognize. While ectopic parathyroid adenomas are thought to be the most common cause of persistent hyperparathyroidism following neck exploration, a number of reasons other than ectopic tumors can be responsible for persistent hyperparathyroidism, such as disease recurrence, surgical inexperience, missed or residual tissue, parathyromatosis and supernumerary glands. Given these challenges, localization studies are recommended prior to reoperation , 2.99mTc-sestamibi scan. If localization remains unsuccessful, cross-sectional imaging is employed (e.g. CT or MRI). Selective venous sampling for parathyroid hormone levels is done only after previous unsuccessful parathyroid exploration. 11C-methionine PET scanning has been very useful in localizing parathyroid adenomas that have failed localization by other techniques (data not yet published). Intraoperative parathyroid hormone monitoring and frozen section are useful at the time of re-exploration.Previous reports in the literature describe an algorithm for the management of persistent hyperparathyroidism , 20. Sim99mTc-sestamibi scan and ultrasound are used as first-line imaging modalities [99mTc-sestamibi scan, CT, MRI, PET-CT, and PTH selective venous sampling were all utilized. PET-CT, 99mTc-sestamibi scan, MRI and even the initial CT scan failed to localize this ectopic adenoma. Invasive techniques such as selective venous sampling are typically reserved for cases where non-invasive imaging has failed to yield an answer; however, this case highlights the potential utility of such methods. Details regarding the specific utility and pitfalls of these various imaging modalities are described elsewhere [The case illustrates several points regarding diagnostic imaging for hyperparathyroidism. Most commonly, dalities . CT and lsewhere .This case demonstrates some of the challenges of surgical management of persistent hyperparathyroidism. In diagnostically challenging cases of persistent hyperparathyroidism, unusual ectopic locations of parathyroid adenomas must be considered. Such consideration is necessary in order to avoid numerous re-operative explorations and the associated risks to the patient. Instead of the upper range of parathyroid glands being stated as the angle of the mandible, we suggest that the parathyroid adenomas should be considered possible at least as high as the nasopharynx. Given the high ectopic potential of these glands, the upper limiting position may not yet be known.


Here we report a very rare entity of an infratemporal region aggressive fibromatosis in a 23-year-old Tharu man who had presented with the symptoms of painless but progressive trismus.We describe a case of aggressive fibromatosis in a 23-year-old Tharu man. Radiological imaging as well as an immunohistochemistry panel from a biopsy indicated a diagnosis of an aggressive fibromatosis. Since there was no aggravation in his trismus following surgery and because of his poor socioeconomic status, he was advised to attend regular follow-up visits without any adjuvant therapy.This case report adds to the notion of keeping the differential diagnosis of an aggressive fibromatosis in all patients presenting with progressive but painless trismus. The characteristic imaging findings as well an immunohistochemistry panel will help us clinch the correct diagnosis. Aggressive fibromatosis (AF) is a very rare entity . Its oriHere we describe a case of AF in a 23-year-old Tharu man; diagnosis followed detailed radiological imaging as well as an immunohistochemistry (IHC) panel. This case report highlights the importance of considering this lesion for any patient presenting with progressive, although painless, trismus with radiological imaging revealing loss of musculoaponeurotic planes within the infratemporal fossa along the pterygoid muscles. IHC panels aid in differentiating it from its malignant counterpart: fibrosarcoma. Our case was also unusual because it involved the infratemporal region, pterygoid muscles, and invasion of the right maxillary sinus.A 23-year-old Tharu man from a remote village in Nepal presented to our out-patient clinic with a history of progressive difficulty in opening his mouth for the last 2 months. He had significant weight loss in that period because of the restrictions in taking oral feeds and chewing them. His symptom was not associated with any pain. There was no history of trauma, fever, joint pain, dental pain, rashes, or any other swellings associated with the symptom. His medical history was positive for having been on anti-tubercular therapy (ATT) for suspected abdominal tuberculosis for the last 3 months. There was no significant past surgical or any relevant positive family history. A local examination did not reveal any visible swelling or any features of inflammation around his temporomandibular joint (TMJ). An intra-oral examination was also normal. His mouth opening was of only one finger width. There were no palpable lymph nodes in his body. A general systemic examination was normal.An X-ray of his TMJ was normal Fig. . ComputePostoperatively, his mouth opening improved to three finger width. He also started gaining weight owing to the ease in eating and chewing.Histology revealed dense fibrocollagenous tissue with moderate to heavy lymphoplasmacytic infiltrates with entrapped muscle with cells of small nuclei arranged in a linear pattern Fig. . There wThe final diagnosis of AF (desmoid tumor) was eventually made. Our patient had been advised to start weekly methotrexate therapy but he denied it citing their poor economic status.He has been on regular follow up with no deterioration in his symptoms in the last 4 months.AF is a rare entity constituting only 0.3% of all tumors with an annual incidence of around 3 to 4 per million of population . Such a Radiological divisions into superficial and deep variants have been made . In MRI The pivotal issue in such a lesion is to differentiate it from its malignant counterpart of fibrosarcoma, which may be both clinically and radiologically challenging . AnotherAlthough surgery is the mainstay of management, safe resection is most often extremely challenging because of the locally invasive nature of AF and its tendency to insinuate into surrounding vital structures. This locally infiltrative growth pattern results in subtotal excision and thereby results in a higher propensity for subsequent recurrence . IntraopTo minimize recurrence of AF, the role of radiation, especially in recurrent lesions, at a dose of 54 Gray has been advocated . To miniThere is also the role of pharmacotherapy using both cytotoxic as well as non-cytotoxic agents in managing such a case in a patient-tailored manner .In selective cases, in which the symptoms are not escalating and repeat imaging shows non-progressive lesions, regular follow up can also be justified .Our case was unusual in the sense that there was involvement of the infratemporal fossa with loss of pterygoid muscle planes as well as invasion to the maxillary sinus. Although the lesion was locally invasive, there has been no aggravation in his clinical symptoms in the last 4 months of his follow-up visits.Although AF is rare, this case report adds weight to the importance of considering AF in the differential diagnosis of any cases presenting with a painless but progressive trismus. Histology and a detailed IHC assay are the cornerstones in arriving at a diagnosis. There is still a major debate about the optimal therapeutic management of such cases; however, it is prudent to take a tailor-made approach on a case-by-case basis.


We report a rare case of an 18-month-old female with autosomal recessive polycystic kidney disease, Caroli syndrome, and pure fetal type hepatoblastoma. The liver tumor was surgically resected with no chemotherapy given. Now 9 years post resection she demonstrates no local or distant recurrence and stable renal function. Hepatorenal fibrocystic diseases (HRFCDs) are a group of severe, monogenic disorders characterized by the common pathological appearance of developmental abnormalities mainly involving the liver and kidneys . Autosom2, normal liver enzymes, and coagulation factors. Serum alpha fetoprotein (AFP) level, checked because of her firm palpable liver, was 500 ng/ml . Her platelet count was 250,000. She was diagnosed with ARPKD and CHF, based on clinical presentation and imaging. Blood pressure was checked as part of the evaluation for renal dysfunction and found to be elevated, despite no symptoms. Endoscopy was not performed. Genetic testing demonstrated two mutations associated with PKHD-1: a novel missense mutation c.1616T>C (p.Ile539Thr) in exon 18 and a frameshift mutation c.3761delCCinsG (p.Ala1254fs) in exon 32.A 2-month-old infant presented with hepatosplenomegaly. Sonographic imaging showed large kidneys with multiple cysts, multiple liver cysts, splenomegaly, and no ascites. Her laboratory results included a creatinine clearance of 43.33 ml/min/1.73 mAt 18-month follow-up, her AFP level rose to 800 ng/ml and an abdominal sonogram demonstrated a new 4.5 cm hyperechoic mass in the right lobe of the liver with involvement of the caudate. She was offered liver biopsy and primary chemotherapy (in her native country). Primary resection was not pursued due to concerns that after resection residual hepatocyte mass would be insufficient to maintain adequate liver function.2 before and 47.5 ml/min/1.73 m2 after the surgery. Histopathology of the resected specimen revealed pure fetal epithelial-type HB with low mitotic activity  to 1.6 (2002–2009) per 1 million in children younger than 19 years (75–1983 tAutosomal recessive polycystic kidney disease and GCKD are part of a clinical spectrum known as HRFCDs; these conditions are linked to genetic mutations resulting in developmental abnormalities of the liver, kidneys, lungs, bones, and central nervous system . ARPKD hThe incidence of concurrent ARPKD, CHF, and HB is rare making universal screening seem cost ineffective. Unfortunately, there are no clinical clues to the development of HB in this setting other than serial AFP assessments and imaging. However, early detection as in our case could permit an aggressive surgical approach sparing precious remaining kidney reserve from chemotoxicity and liver transplantation. So, the benefit of screening to the individual may justify the costs involved.Although the progression of renal and liver diseases varies, the majority of the patients with ARPKD will develop renal dysfunction requiring renal replacement therapy and ultimately transplantation. Cisplatin is one of the most active chemotherapy agents for HB  but is pNevertheless, this approach should be reserved for selected patients who qualify for primary surgery based on PRETEXT classification. We believe that these patients should be referred to centers with surgical expertise in tumor management and solid organ transplantation. Complications of hepatic surgery in children with HB are associated with significant morbidity and mortality rates, reducing the 5-year overall survival from 72 to 50% in high-risk groups , despiteThe surgical approaches to HB differ between European and North American centers. The Société Internationale d’Oncologie Pédiatrique—Epithelial Liver Tumor Study group, representing European centers, have used a single strategy for all patients including neoadjuvant chemotherapy followed by surgical intervention regardless of size or histology, in order to make the tumor smaller, more delimited from the surrounding hepatic parenchyma and more likely to be completely resected. In contrast, the children’s oncology group, representing North American centers have offered resection at diagnosis whenever prudently feasible followed by adjuvant chemotherapy as a strategy to minimize overall chemotherapy exposure and toxicity . The decThis case represents a successful approach for the treatment of an unusual combination of HRFCD and HB. The rarity of this association warrants this report and merits meticulous genetic investigation to discover factors predisposing to both entities. The case also illustrates a personalized treatment approach based on a particular clinical situation taking into account all organ systems involved and resulted in an excellent outcome compared to similar cases in the literature treated with traditional approaches. Standardized approaches to specific tumors remain prudent; however, alternative therapies in patients with malignancies and impaired renal function taking into account the nephrotoxicity of the treatment regimen merit equal consideration. Although no formal recommendation can be drawn from a few case reports, we feel that given the reported association, screening for HB using AFP and imaging of the liver could be considered in patients with ARPKD and CHF, to warrant early detection of tumor. This practice could permit an individualized approach using upfront resection and elimination/minimization of chemotherapy.This is a case report and does not require committee approval. The authors communicate with the parents of the minor and got verbal consent to publish this report. This report does not include any identifiers of the patient to protect patient confidentiality.All the authors made a great effort to build this novel case report. NK being the first author, constructed a draft based on the patient’s case presentation and history. SL, NO, HR, DY, and JE added the literature review in the draft and made necessary changes in the draft accordingly, making it more concise. MM, being the corresponding author design the case report, supervise each step during the writing of this case report, took a remarkable effort to enhance the quality of the manuscript since the beginning, and made the final assessment as well.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Malignant Deciduoid Mesothelioma (MDM) is an extremely rare variant of epithelioid mesothelioma. It was first described in young females, in the peritoneum, and its relation with asbestos was not well defined.Later reports, have shown that this variant may also occur in the pleura, the pericardium and the tunica vaginalis of elderly people, who had been exposed to asbestos.We report a case of malignant deciduoid mesothelioma that occurred in the peritoneal cavity, and the omentum of a 35-year-old woman. The patient had never been exposed to asbestos.Through this observation, we describe clinical, histopathological, and immunohistochemical findings of deciduoid mesothelioma, and review the literature reports. MDM is an extremely rare subtype of malignant epithelioid mesothelioma, with a poor prognosis. It was first described by Talerman et al.  in 1985 Few cases have been reported in the literature to date, in the form of single case reports. A total of 25 cases of deciduoid peritoneal mesothelioma have been documented in the English literature, since this entity was first described in 1985 –17.The aim of the present report is to describe MDM of the peritoneum, to discuss the differential diagnosis and management of this extremely rare tumour.A 35-year-old female patient, with no medical history, was admitted for an appendicular syndrome. There was no history of asbestos exposure. On physical examination, she had a low-grade fever. A generalized abdominal tenderness and distension were also reported. Abdominal ultrasound examination showed moderate ascites, and hyperechogenic mesenteric fat. An exploratory laparotomy revealed multiple firm nodules, white to gray, infiltrating the peritoneal cavity, and the omentum. A biopsy was performed. We received four firm, gray white fragments. Microscopically, H&E stain showed sheets of malignant cells, formed exclusively by large pleomorphic epithelioid cells with abundant, eosinophilic cytoplasm. The nuclei were large and vesicular, with prominent nucleoli Figs.  and 2. TIn immunohistochemistry, tumor cells were positive for cytokeratin AE1/AE3, calretinin , melanoma and epithelioid angiosarcoma [Positive staining for AE1/AE3 and calretinin are useful to exclude Rhabdomyosarcomaosarcoma , 21. ImmChemotherapy and cytoreductive surgery are usually the best therapeutic options. Cisplatin and pemetrexed have demonstrated a significant survival advantage, and are considered as firstline treatment , 17.The deciduoid subtype was regarded by some authors to be a more aggressive variant . HoweverIn summary, through this observation, we want to highlight the rarity of this variant. It is important for pathologists to recognize this morphological variant of epithelioid mesothelioma, easily confused with other diangnosis, and discuss the available treatment options. We believe that more studies on a larger number of cases may be helpful in further defining this entity.


Cemento-osseous dysplasia is a jaw disorder characterized by a reactive process in which normal bone is replaced by connective tissue matrix. There are different Cemento-osseous dysplasia entities. The treatment of these lesions, once diagnosed by radiology, is not required because generally they are asymptomatic. The localization is in the tooth-bearing areas of the jaws and its distribution is symmetric.In this case report, a 57-year-old Caucasian female patient was referred to our attention complaining of painful inflammatory events localized in the right angle of the jaw. The radiographic appearance, the distribution of several lesions and the positive vitality test of the involved teeth, supported the diagnosis of Florid Cemento-osseous dysplasia. Because of the symptomatology, the patient was submitted to surgery and the lesion and the second inferior right molar were removed. The histological examination of the specimens confirmed the diagnosis.Many lesions that may exhibit a similar sclerotic appearance on conventional radiographs have to be differentiated and dental imaging can be used to discriminate between Florid COD and other lesions. Diagnosis of Florid Cemento-osseous dysplasia can be made with accurate clinical and radiographic assessment. In asymptomatic cases no treatment is required and the patient should have regular follow-up, but in this symptomatic case it was necessary to proceed with surgical intervention. The surgery treatment in the symptomatic case had a favourable prognosis and the two years follow-up has shown a complete healing. Given the abow, it is concluded that the choice of treatment must be selective according to the disease sites. Key words:Cemento-ossifying dysplasia, fibro-osseous lesions, florid cemento-osseous dysplasia, cementoma.  Fibro-osseous lesions of the jaws are of particular interest to oral and maxillofacial surgeons, radiologists and pathologists, as they emphasize the crucial role of these specialists in the diagnostic process and therapy. The differential diagnosis is important because misdiagnosis may lead to unnecessary endodontic treatment, incisional biopsy or surgical removal. The term fibro-osseous lesion (FOL) is a generic designation of a group of jaw disorders ,2.In these lesions, bone is replaced by benign connective tissue matrix that displays varying degrees of mineralization in the form of woven bone or of cementum-like round acellular intensely basophilic structures indistinguishable from cementicles ,4. The tCOD represents a reactive process in which normal bone is replaced by a poorly cellularized cementum-like material and cellular fibrous connective tissue. They are classified, depending on clinical and radiographic findings, into three subtypes: periapical (limited to the anterior mandible), focal (single lesion), and florid .These lesions, that present an atypical radiographic appearance, require a detailed clinical and laboratory workup to arrive at a correct diagnosis in order to develop an appropriate treatment ,10.On radiographic evaluation, during the early stage, a COD lesion may be detected as a round or oval apical radiolucency with a well-defined radiopaque border. In the second mixed stage, a radiolucent lesion may include radiopacities. In the mature stage, the internal mixed area becomes completely radiopaque with a thin radiolucent periphery .Commonly, no treatment is required and only regular follow-up examinations are recommended ,13.In the 2005 edition, the World Health Organization (WHO) classification of odontogenic tumors, classified osseous dysplasia into four groups based on the location .•Periapical Osseous Dysplasia (PCD)•Focal Osseous Dysplasia •Florid Osseous Dysplasia•Familial Gigantiform Cementoma The term “florid” related to cemento-osseous dysplasia was introduced to describe the extensive manifestations of the disease in multiple quadrants of tooth-bearing area of the jaws ,16. The FCOD is usually asymptomatic and diagnosed accidentally at routine dental radiographic examination. Dull pain, drainage, exposure of the lesion in the oral cavity, focal expansion and facial deformities are present when infection occurs ,21. RadiIn 2007, a 49-year-old Caucasian woman was referred to our ward for a routine dental check up. Her past medical history was not notable, and there was no evidence of systemic disease.On intraoral examination, the patient was asymptomatic, there was no swelling of the oral mucosa and all present teeth were vital except the second upper left molar. A panoramic X-Ray showed different apical lesions. In the jaw the apical area of incisors and the edentulous area of the right third molar were involved. In this latter area, the lesion extended mesiodistally from the distal root of the second molar and from the crest of the edentulous alveolar ridge to the inferior alveolar canal. The lesion had a somewhat multilocular appearence, a radiolucent area surrounded radiopaque structures, and the bone adjoining the borders of the lesions was sclerosed, giving a pseudo-corticated appearance. There was no root resorption or fusion of the lesions to the involved teeth. In the maxilla we could observe radiolucent lesions apically at the second right incisor and at the first left premolar. Because of the asymptomatic nature of the lesions, no further treatments were proposed. The patient was advised to have a routine follow-up examination six-months later, but she returned only in 2015 complaining of recurrent episodes of abscesses and exhibiting a significant degree of mobility of the second right molar. During this period the patient did not attend regular recall visits a tour department and she underwent endodontic treatment alio loco. She recalled the use of antibiotics without specifying type and duration.A new panoramic radiograph Fig.  showed aOn March 2014 a CT examination was performed in both jaws. In the vertical reconstructions of the maxilla multiple patchy well defined hyperdense lesions with hypodense rim located around root apices of both second incisors and the first left premolar were present. A sclerotic margin surrounded these lesions. Lesions above both lateral incisors were connected with buccal cortical bone without interruption. In the axial view we could observe the lesion that surrounding the first left premolar extended to the lingual cortical, no interruption. Panoramic-like reconstructions demonstrated the symmetry and the mesio-distal extension of these lesions. There were no root resorptions in any case. In the mandible multiple patchy well defined hyperdense lesions with hypodense rim and sclerotic borders located around root apices of the lower incisors were present, similar to the upper lesions in the maxilla. These lesions were in contact with the lingual cortical plate with no interruption of it. In the edentulous area of the right third molar the lesion appeared amorphous, lobulated and mixed radiolucent and radiopaque masses (cotton-wool appearance) with sclerotic borders. In the axial view the lesion looked something like an impacted tooth. Inferiorly to the second molar another radiolucent lesion that eroded the buccal cortical plate was present. The panoramic view showed that there were not interruptions of the inferior alveolar canal.FCOD lesions that are not infected do not require any treatment procedure, but because of the septic complication and the symptomatology, a decision was made to extract this molar, to enucleate the lesion and to do histological examination. After the completion of the clinical and radiographic examination, the patient was scheduled for surgery under general anesthesia.During surgery, a intrasulcular buccal incision of the second right mandibular molar was performed with a mesial extension to the distal side of the first right mandibular molar and a mucoperiosteal flap was raised. After the extraction of the second right inferior molar, the apical lesion was enucleated Fig. . This leThe patient presented for radiography control 6 months after surgery. The patient was called back to our department 2 years after surgical treatment for a new radiographic follow-up that shows a complete healing Fig. .Our diagnostic hypotheses, based on medical history, clinical and radiological information was Florid Cemento-Osseous Dysplasia. Histological results show “ginger root” bony trabeculae surrounded by connective tissue matrix highly cellularized. There are bony trabeculae surrounded by activated osteoblast and osteoclast. At this point, diagnosis is made with clinical-pathological relationships:•Focal cemento-osseus dysplasia is present if the posterior quadrant is involved•Periapical cemento-osseus dysplasia is present if it involves an anterior quadrant •Florid cemento-osseus dysplasia is present if it occurs in more quadrantsAll of these clinical situations share the same histological features including cemento-ossifyng fibroma from which they only differ by the lack of a capsule.Etiology of cemento-osseus dysplasia and cemento-ossifying fibroma is not yet clearly understood, but the close proximity to the periodontal ligament may lead to the hypothesis that both cemento-osseus dysplasia and cemento ossifying fibroma originate from the same tissue. Furtherly, cemento-osseus dysplasia may represent a reactive lesion, whereas fibroma looks like a neoplasm. The former is developed apically to the root or on a site of tooth extraction , whereas COF often did not exhibit association with teeth but causes root displacement without root resorption.Radiologically, the radioopaque aspect in Focal Cod is irregular or diffuse like “cotton wool”, otherwise radiodensity in COF is smoothly distributed like “snow flakes”. The explanation is the histopathological exam, “cotton wool” represents “ginger root” bony trabeculae in Focal COD, in contrast “snowflakes” appearance may reflect the bony trabeculae seen in COF . RegardlIn the presented case radiological lesions show a “cotton wool” aspect which is also typical of Paget’s disease, but the latter usually affects the entire mandible in addition with a loss of the lamina dura . Paget’s disease often involves other bones such as the spine or femur and causes biochemical serum changes as elevated alkaline phosphatase levels, which on the other hand is within normal limits in patients with FOCD ,25.A further radiopaque lesion that appears with even greater frequency is idiopathic osteosclerosis (IOS) but it has no radiolucent periphery, that otherwise is present in COFs, FD, odontomas and in our case and even it causes no expansion ,27. MoreAmeloblastoma is a benign tumor. It originates from odontogenic epithelium and is characterized by painless slow-growing swelling. Generally it occurs in the mandible, especially in the posterior area and ascending ramus, producing root resorption. We have not seen these characteristics in our case.About FCOD management, it is reasonable to keep asymptomatic patient under observation. In the absence of clinical signs, it would be recommended a revaluation with panoramic radiograph every 2 or 3 years. When the patient is symptomatic the management becomes more difficult because chronic inflammation and infection develop within densely mineralized tissue. In this case, antibiotics have poor tissue diffusion and generally are not effective. Therefore, biopsy increases the risk of infection and it’s not reasonable to remove these lesions in asymptomatic patients ,30.Florid Cemento-osseous dysplasia needs an accurate clinical and radiographic assessment to be diagnosed. In the asymptomatic cases treatment is unnecessary, the patient should have a regular follow-up to check any changes. However, in the aforementioned case, since it was symptomatic, and considering that it was evolving in a chronic infection due to diseases of the adjacent teeth, it has been necessary to do surgery to solve the algic dysfunctional condition. Infact, the radical surgery treatment in the symptomatic cases has a favourable prognosis and allows a complete healing. The choice of this treatment should be selective according to the disease sites. The goal of this work is to highlight that symptomatic cases of bacterial FCOD overlap of maxillary bones, can not be solved with drug therapy alone, radical surgery leads to a recovered fully demonstrates clinically and with radiology.


Synovial osteochondromatosis (SOC) is a benign tumor characterized by synovial connective tissue metaplasia. SOC commonly affects major joints including the knee followed by the hip, elbow, and wrist. SOC cases in the hand are not reported as often as SOC of major joints. Particularly SOC of the carpometacarpal joint of the thumb is rare. We report on a 57-year-old female with primary SOC of the carpometacarpal joint of her left thumb. Surgical excision was performed and the patient had no symptoms with full range of motion of her left thumb. At 3 years of follow-up, there was no recurrence. Synovial osteochondromatosis (SOC) is a benign neoplasm characteristic of synovial connective tissue metaplasia within the synovial membranes of joints, tendon sheaths, or bursae –3. SOC cA 57-year-old woman was present at our institution with a chief complaint of the pain and swelling at the left thumb. She had the pain for 5 years and noticed the swelling 3 years ago. She had no history of trauma and no other relevant medical history. On physical examination, the patient had a palpable and tender mass, approximately 2.5 cm × 2.5 cm × 1.5 cm in diameter, located over the dorsal aspect of the CM joint of the left thumb without involvement of the skin . ResultsA plain X-ray of the hand revealed mild soft tissue swelling and radiopaque bodies around the CM joint of the left thumb without erosion of the base of the first metacarpal bone and trapezium . ComputeMarginal resection of the tumor with synovectomy was performed after the CM joint capsule was incised Figures  and 5. PThere is no evidence of recurrence and the patient had a return of full range of motion of her left thumb 3 years postoperatively.This was a rare case of SOC in the CM joint of the thumb. SOC is a benign tumor characterized by a metaplasia of the synovial membrane resulting in an accumulation of intra-articular cartilaginous nodules –3. The eThe most common articular lesion of SOC is the knee followed by the hip and elbow , 5. SOC Symptoms of SOC are nonspecific, including a palpable mass, swelling, pain, tenderness, and restricted movement of the joint , 4. CompFor making a diagnosis histological examination is necessary. The common characteristics of SOC on gross pathology are synovium with multiple nodules. SOC on microscopic examination is known to have focal and circumscribed areas of hyaline cartilage embedded within synovial connective tissue . The difThe most effective treatment is synovectomy with removal of loose cartilaginous nodules , 14. RecSynovial osteochondromatosis in the carpometacarpal joint of the thumb is rare. Surgical excision and histopathological examination are necessary for diagnosis.


Hepatocellular carcinoma (HCC) is the commonest primary malignant neoplasm of the liver in most countries with a notoriously poor prognosis. Variation in global incidence is well-recognized and the occurrence of HCC is linked to several established environmental, dietary, and lifestyle factors. HCC demonstrates morphological heterogeneity both within the same tumor and from patient to patient. Differing architectural patterns and cytological variants may be seen. Inclusion bodies are believed to represent organized structures of proteins which contribute to their pathogenesis and share several constituents like chaperones, p62, ubiquitin, and Valosin containing protein. The various hepatocyte cytoplasmic inclusions described in HCC include Mallory-Denk bodies (MDBs), hyaline bodies (HBs), glycogen, fat, fibrinogen, alpha 1 antitrypsin (AAT), and ground glass. MDBs are the most common inclusions seen in hepatocellular carcinomas. The two cases shared intracytoplasmic inclusions which are characterized by larger sizes and present in every section examined. These exhibited features of MDBs and HBs present in most tumor cells, further supporting close relationship. The first patient is an 83-year-old man who presented with vague upper abdominal discomfort, weight loss, and general weakness. Blood work was normal and upper and lower gastroscopy revealed no specific lesion or malignancy.A solitary lesion in the left lobe of liver was found on computed tomography (CT) scan. Tumor markers were noncontributory and hepatitis serology was negative.He has a medical history of hypertension controlled with antihypertensive medications. Liver biopsy showed hepatocellular carcinoma. Surgical resection of segments 2 and 3 including the mass was performed.The second patient is a 56-year-old female who presented to the emergency department with abdominal pain. Medication used included oral contraceptive and hormonal supplements. CT scan showed 11 cm heterogeneous liver mass in the right lobe with evidence of intralesional hemorrhage and associated contrast extravasation requiring urgent surgical intervention. The lesion was resected. Hepatitis serology result was negative.Macroscopic examination of the first lesion showed a partial hepatectomy with 4 cm lobulated green/yellowish mass with surrounding nodular and fibrotic liver parenchyma. The second lesion consisted of an ill-defined tumor, 13 cm size, with a yellow/brown cut-surface and a small peripheral rim of grossly normal liver parenchyma .Microscopically, both cases showed HCC with admixed trabecular and pseudoacinar patterns. The malignant hepatocytes were enlarged and contained prominent, densely eosinophilic intracytoplasmic globules, many of which had surrounding clear halos admixed with Mallory hyaline bodies (arrow) Figures  and 3.The globular structures were negative for Masson's trichrome, amyloid, and Periodic Acid Schiff/Periodic Acid Schiff Diastase (PAS/PASD) special stains.By immunohistochemistry  the intracytoplasmic globules showed expression of p62 and focal positivity for ubiquitin Figures .CAM5.2 showed very focal, weak expression. Fibrinogen, alpha-fetoprotein, AAT, alpha 1-antichymotrypsin, and fibrinogen immunohistochemistry were all negative. The morphology and predominant CAM5.2 negativity in the majority of the inclusions suggest they are mainly HBs while the focal, weak CAM5.2 staining indicates a focal MDB component.Case one had occasional neoplastic hepatocytes that also contained cytoplasmic fat and was associated with changes of nonalcoholic steatosis in the adjacent liver.The background hepatic parenchyma in case two demonstrated changes consistent with cirrhosis.Electron microscopy revealed dense fibrillary material arranged in parallel with occasional vesicular structures present.Hepatocellular carcinoma (HCC) is the commonest primary liver cancer in most countries with a recognized global variation in incidence, being most prevalent in China, Southeastern Asia, and Sub-Saharan Africa . There iChronic viral infection (HBV or HCV), alcohol induced liver injury, ingestion of high levels of aflatoxin, iron overload (hereditary hemochromatosis), and alpha 1 antitrypsin deficiency are amongst the accepted risk factors for HCC. The tumor commonly arises in cirrhotic livers.Symptoms may be due to the tumor or the predisposing chronic liver disease and include abdominal pain, general fatigue, anorexia, weight loss, nausea, or vomiting.Microscopically, HCCs show loss of the normal reticulin network with widening of the cell plates (>3 cells thick). The tumor cells resemble hepatocytes to a variable extent, depending on the degree of differentiation. The presence of intercellular bile and identification of canaliculi confirm the hepatic cell lineage. They may show small or large cell change and be associated with cytological atypia, mitotic activity, and “capillarisation” of the sinusoids.The report described two cases which presented to the emergency with abdominal pain. On CT scanning both patients showed single hepatic mass. No past history of liver diseases was noted and serology testing for viral hepatitis was negative. The initial clinical diagnosis for the first case was metastatic carcinoma and for the second case it was hepatic adenoma. Following proper clinical assessment both patients underwent surgical resection of the masses which revealed HCC with florid intracytoplasmic inclusions.Intracytoplasmic inclusions are not uncommon or specific to HCC. MDBs, first described by Mallory in 1911 , are theDenk et al.  demonstrThe inclusions vary in sizes and can be few or numerous with zonal distribution. Special histochemical and immunohistochemical markers are required to assist in delineating some of the other inclusions such as AAT globules and fibrinogen.Many of the hepatocyte intracytoplasmic inclusions are not specific for any disease entity but identifying a particular type of inclusions in the appropriate histological and clinical setting can point to a particular diagnosis.The prognosis of HCC is a controversial  issue; hWe report two cases of HCC with unusually florid large, prominent intracytoplasmic inclusions with features of HBs and MDB present. This is in support that both inclusions share common molecular constituents and pathogenesis.