Vascular leiomyomas or angioleiomyomas are rare tumors that can be found in the nasal cavity. The etiology of angioleiomyoma remains poorly understood and there are several hypotheses to explain the origin of sinonasal leiomyoma. We here describe the clinical and histological findings in a case study along with the feasibility of surgical treatment using a radiofrequency instrument. In particular, we describe the case of an adult patient with recurrent epistaxis because of a nasal angioleiomyoma and the performed treatment in the form of complete surgical excision. Radiological imaging is a helpful tool to give an indication of the extension of the tumor, as well as for the proper planning of the surgical approach. Either MRI or CT scans are found to be best suited for this purpose. This case report recommends the complete surgical excision of the angioleiomyoma, by either an endoscopic or an open procedure. This can be safely performed using a radiofrequency instrument as shown in this case with no recurrence during a follow-up of 12 months. Vascular leiomyoma (VL) or angioleiomyoma comparable to the one described in this study is extremely rare. Only 8.5% to 10% of VL are found in the head and neck area and, among these, only 3% occur in the nasal cavity . IntranaA 45-year-old man reported to his general practitioner with recurrent and strong, but self-limiting, left-sided epistaxis over the preceding 2 to 3 months, each time lasting up to 10 minutes. He also felt an intermittent, dull pain on the left side of his nose and the strange sensation of a slowly growing mass inside his nose, but neither nasal obstruction nor hypesthesia of the face. An excision under local anesthesia was then attempted. The effort was abandoned because of strong bleeding, a nosepad was applied and then the patient was referred as an emergency to our clinic. Upon arrival the bleeding had stopped.The personal history showed an appropriately treated arterial hypertension and no regular alcohol or nicotine consumption or substance abuse. There was no high incidence of cancer in the family history.A clinical examination showed a healthy albeit obese man with a marginally elevated blood pressure level. After removal of the nosepad and suction cleaning, the nasal inspection showed a well-defined tumor of the lateral nasal wall on the anterior face of the inferior turbinate, approximately 10 mm in size. The tumor surface exhibited several incisions. Anterior and posterior rhinoscopy revealed a slight deviation of the nasal septum, but no other intranasal pathology. The external nose showed a slight, though distinctly palpable swelling in the area between the lateral cartilage and the nasal bone on the left side.W × H × D) sized, nodular, contrast enhancing lesion emanating from the lateral nasal wall and extending to the anterior face of the inferior nasal turbinate on the level of the piriform aperture without any sign of infiltrative growth.To further investigate the repeatedly bleeding endonasal mass a CT scan with contrast was conducted. It showed a tumor of the inferior turbinate, only marginally contrast enhancing, 24 mm in the largest diameter. No bony erosion was reported, and an infiltration of the lateral nasal cartilage could not be ruled out . A biopsA transnasal endoscopic tumor resection was performed under general anesthesia using a radiofrequency instrument (Ellman Surgitron) . IntraopVascular leiomyomas are benign neoplasms that are rarely found in the head and neck region. The presenting symptoms are nonspecific and usually characterized by nasal obstruction, epistaxis, and nasal discharge. Local pain is not common in sinonasal leiomyoma but has also been described. Vascular leiomyomas occur as small, painless masses in the superficial layer of the skin or mucosa. Auricle, nose, lip, and neck are the most common areas of occurrence .These tumors can affect persons of any age, but they are more common in people between 30 to 60 years of age with a female predominance  .While the etiology of sinonasal leiomyoma remains uncertain, there are three dominant hypotheses to explain the origin of sinonasal leiomyoma.They could stem from aberrant undifferentiated mesenchymal or smooth muscle elements in the blood vessel wall . In the Some authors suggest that vascular leiomyoma could be a vascular malformation or a progressive development of smooth muscle proliferation from hemangioma, to angioma with much muscle, to leiomyoma with many vessels, and to solid leiomyoma . The infMarioni et al.  hypothesThe World Health Organization (WHO) classified leiomyoma into three groups: nonvascular leiomyoma, vascular leiomyoma, and epithelioid leiomyoma. Morimoto divided angiomyoma into three histologic subtypes in 1973: solid or capillary, cavernous, and venous. The vascular subtype is the most common one and angiomyoma of the head and neck usually occurs as a venous or cavernous type . The solThe histopathological differential diagnosis of angioleiomyoma includes hemangioma, angiofibroma, myopericytoma, fibromyoma, and leiomyosarcoma . SinonasInverted papillomas and osteomas are the most common benign tumors followed by fibrous dysplasia and neurogenic tumors, for example, schwannomas.Squamous cell carcinomas are the most common malignant sinonasal tumors (80%), followed by adenocarcinoma. Chondrosarcoma, lymphoma, salivary gland tumors, neuroendocrine tumors, and mucosal malignant melanoma represent a further differential diagnosis.Radiological imaging, for example, MRI and/or CT scans, represents an helpful tool for visualizing the extension of the tumor and for the proper planning of the surgical approach, but there are no particular imaging techniques that specifically characterize the VL . NeverthThere are no reports of recurrence after total excision , 15. YanAngioleiomyoma in the nasal cavity is extremely rare but an important differential diagnosis in a patient with a nasal tumor. Complete surgical excision is recommended either by an endoscopic or by an open procedure. Attention should be paid to intra- and postoperative hemorrhage due to the hypervascularity of this neoplasm. In our case of a VL originating from the anterior border of the inferior turbinate, we demonstrated that the endoscopic approach with a radiofrequency instrument resulted in a complete and safe removal of the mass with no recurrence during a period of 12 months.


Abdominal bloating is a common clinical presentation and can impact significantly on the quality of life. It can be functional or signify more serious pathology.We present a case presentation of a 38-year-old man who presented with recurrent episodes of abdominal bloating and severe debilitation for many years. He had suffered weight loss and was unable to tolerate solids. Ten years ago, he underwent an emergency laparotomy for sigmoid volvulus. In the presence of anaemia, normal imaging, and normal endoscopy, a laparoscopy was performed. At surgery, several adhesional bands were identified and resected. He had an uncomplicated recovery.Acute and chronic adhesional bowel obstruction are common presentations and carry a significant morbidity and mortality. Elective laparoscopy can be a valuable tool for diagnostic and treatment purposes. Pathology should be suspected in cases where weight loss is a feature. Abdominal bloating is a common patient presentation, estimated to affect ~30% of the general population -3 and caA 38-year-old gentleman presented to the surgical clinic as a physician referral. He had been suffering for eight years with frequent episodes of abdominal bloating, nausea, weight loss, and constipation. Every time he consumed solid food his abdomen would distend to such an extent he would have to retire to bed until the episode settled. He would then pass a large amount of gas and his abdomen would deflate. He had been on a fluid diet for the last twelve months but had not been admitted to hospital during this time. His bowel habit was irregular and had not noticed blood in his stool. Of note, he had undergone an emergency laparotomy ten years ago for sigmoid volvulus whilst on holiday abroad, although he was not aware of exactly what operation had been performed, and no paperwork pertaining this was available. He was otherwise fit and well and took no regular medications. He had no family history of cancer or inflammatory bowel disease. On examination, he was cachectic with a well-healed midline laparotomy scar. He had no hernias and rectal examination was normal.Full blood count revealed a normocytic anaemia (Hb10.9), and urea, electrolytes, and thyroid function were normal. Inflammatory markers were not raised. An abdominal x-ray showed prominent small bowel loops only. Upper and lower gastrointestinal endoscopy were reported as normal, as was an ultrasound and computerised tomography of his abdomen and pelvis.- Functional bowel disorder- Intermittent mechanical obstructionInformed patient consent was obtained for his treatment. This gentleman elected to undergo a laparoscopy during which two adhesional bands were found and resected. Multiple loops of small and large bowel were freed from adhesions to the abdominal wall and to each other Figure . All bowThe patient was discharged home within 12 hours. He successfully ate a bowl of breakfast cereal before leaving the ward, a feat he had previously been unable to perform. He continued to increase his diet at home, and at six-month follow-up had put on weight.The incidence of intra-abdominal adhesions following surgery is estimated at 67-100% -6. They Abdominal distension and change in bowel habit is a common presentation to both physicians and surgeons and, in some cases, can be extremely debilitating for patients. This patient had been suffering from an undiagnosed bowel complaint for eight years. Diagnosis and management can be challenging, and all cases should be approached in a logical fashion. Intolerance of solids and weight loss should alert one to an organic pathology, and should be investigated as a matter of urgency. If symptoms suggesting colorectal cancer are present (see Table Recurrent abdominal bloating is common, and a careful history and examination is paramount. Pathological causes should always be suspected if the diagnosis is uncertain. Adhesional obstruction after laparotomy is frequent, and clinical management depends on the patient presentation. It can be treated conservatively or surgically. Intermittent or subacute small bowel obstruction may not present in a classical way, and a high index of suspicion will aid in diagnosis.


Urethral tumors are rare and aggressive. They usually affect men (2:1) and occur more commonly in white (85% of cases). Soft tissue sarcomas are a heterogeneous group of tumors that arise from embryonic mesoderm. It represents 1% of all cases of urinary tract malignancies and rarely primary affect the ureter. We report a case of male urethral sarcoma. To date, only two similar cases have been published in literature. The epidermoid carcinoma is the most common histological type (80% of cases), followed by transitional cell cancer (15%) and adenocarcinomas (5%). The most affected local is bulbomembranous urethra followed by penile and prostatic urethra.4The disease has an unspecific presentation and, according to some case series reported, the most common symptoms are hematuria, dysuria, urinary retention and urinary incontinence.5Soft tissue sarcomas constitute a heterogenous group of tumors that appear in the embryonic mesoderm and account for 1 to 2% of all malignant disease of the urinary tract.6 The most frequent types of genitourinary sarcomas are leiomyosarcoma, liposarcoma and rhabdomyosarcoma.6In general sarcomas most common forms are liposarcoma, malignant histiocytoma and leiomyosarcoma.8We report a case of sarcoma of the male urethra that, so far, only two similar cases have been reported in the literature.We report a case of 73-year-old white man with history of myocardial infarction (MI), which occurred 3 years earlier, and who was diagnosed with systemic arterial hypertension. He was not a smoker and did not consume alcohol. The patient’s complain was macroscopic hematuria that started intermittent and then continuing that lasted for 1 year.At physical examination no visceromegaly or lymphadenomegaly was observed. The urological exam showed urethrorrhagia without sings of macroscopic lesions. Rectal examination revealed a 30g prostate without nodularity and painless. Patient had normal blood count and coagulogram, early-onset chronic renal failure (urea was 49mg/dL and creatinine was 1.5mg/dL). Total abdominal ultrasound was normal and pelvis examination with contrast showed an extensive injury in posterior urethra that invaded the prostate, bladder and perivesical tissue , but no The patient underwent a cystoprostatectomy with removal of the testicles, pelvic lymphadenectomy and ureteroileostomy using the Bricker method . After tThe anatomopathological study showed a high histological degree pleomorphic sarcoma, measuring approximately 20.0cm, in the bulbar urethra with neoplasia affecting the bladder base, periprostatic region, seminal vesicles, base of the penis and cavernous bodies . The ure9 Prognosis is relatively poor and can be explained by the high proportion seen in high degree tumors, large proportion of patients with metastatic disease, large tumor and the area affected. In addition, the rarity and heterogeneity of genitourinary sarcomas can explain the great variability in clinical progress in different subgroups.10Survival rates of patients with genitourinary sarcomas is worse compared with other soft tissue regions.10Dissemination of urethral cancer follows the anatomic subdivision. The anterior urethra has a lymphatic drainage system for superficial and deep inguinal region. Posterior urethra drains the lymphatic ganglion of external iliac artery, hypo gastric, and internal obturator muscle.10Late diagnosis is seen in one third of patients with inguinal lymphatic ganglion metastasis and in 20% of those with pelvic ganglion metastasis. The most common distant metastasis appears in lungs, liver and bones.10Anterior tumors present better prognosis reaching up to 60% in 5 years. Tumors <2cm have 81% of survival for 5 years. On the other hand, tumors of posterior urethra have 10% survival rate for 5 years if <2cm, 37% of survival for 5 years when presenting tumors 2 to 4cm and only 7% when >4cm. In general, in a stage higher than T2 the survival rate for 5 years is observed in <26%.10Histological characteristics of genito-urinary sarcomas are quite variable. Most of them present high degree of differentiation and isolated focus of squamous metaplasia.7 The second was a 65-year-old man who underwent a perineal uretrostomy to treat a distal urethral stenosis 8, with positive surgical margin in a clinically severe patient with cardiomyopathy that led to an unfavorable outcome. The MI presented by the patient in early postoperative period did not enable the adjuvant therapy with radiotherapy and chemotherapy. It becomes clear that these tumors are extremely aggressive and have poor prognosis. 2Os tumores de uretra são muito raros e bastante agressivos. Acometem mais frequentemente homens (2:1) e são mais comuns na raça branca (85% dos casos).2 A faixa etária do diagnóstico de tumor uretral maligno é de 50 a 79 anos.3São relatados aproximadamente 600 casos de câncer uretral masculino na literatura mundial. O carcinoma epidermoide é o tipo histológico mais comum (80% dos casos), seguido do carcinoma de células transicionais (15%) e dos adenocarcinomas (5%). O local mais acometido é a uretra bulbomembranosa, seguida da uretra peniana e prostática.4A doença tem apresentação inicial inespecífica e, de acordo com algumas séries de casos relatados, os sintomas mais comuns são hematúria, disúria, retenção urinária e incontinência urinária.5Os sarcomas de tecidos moles são um grupo heterogêneo de tumores que surgem a partir da mesoderme embrionária, que representa 1 a 2% de todos os casos de doença maligna urinária.6 Em relação aos sarcomas geniturinários, o tipo mais frequente é o leiomiossarcoma, seguido do lipossarcoma e do rabdomiossarcoma.6Os sarcomas, em geral, possuem como formas mais comum o lipossarcoma, seguido do histiocitoma maligno e do leiomiossarcoma.8Relatamos e discutimos aqui um caso de sarcoma de uretra masculino, que, por sua raridade, tem apenas dois casos relatados.Paciente de 73 anos, do sexo masculino, raça branca, com história de infarto agudo do miocárdico (IAM) há 3 anos e diagnóstico de hipertensão arterial sistêmica, sem história de tabagismo e etilismo. Apresentou-se com queixa de hematúria macroscópica, inicialmente intermitente e, posteriormente, contínua, de início há 1 ano.No exame físico, não foram notadas visceromegalias ou linfonodomegalias. Ao exame urológico, notou-se uretrorragia, sem sinais de lesões macroscópicas. O toque retal revelou próstata de 30g sem nódulos, indolor. O paciente apresentava hemograma e coagulograma normais, função renal com doença renal crônica incipiente . O ultrassom de abdome total era normal, porém a ressonância nuclear magnética de abdome e pelve com contraste evidenciou lesão extensa em uretra posterior, que invadia próstata, bexiga e tecido perivesical , sem alto dia pós-operatório evoluindo a óbito.O paciente foi submetido à cistoprostatectomia com emasculação, linfadenectomia pélvica e ureteroileostomia à Bricker . ApresenO estudo anatomopatológico evidenciou sarcoma pleomórfico de uretra bulbar de alto grau histológico, com neoplasia acometendo base de bexiga, região periprostática, vesículas seminais, base de pênis e corpos cavernosos, com maior diâmetro aproximado de 20,0cm . O tumor9 O prognóstico relativamente pobre pode ser explicado pela maior proporção de tumores de grau elevado, a maior proporção de pacientes que se apresentam com doença metastática, um maior tamanho tumoral e o local anatômico. Além disso, a raridade e a heterogeneidade dos sarcomas geniturinários podem significar grandes diferenças de evolução clínica nos vários subgrupos.10A taxa de sobrevivência de doentes com sarcoma de geniturinário é pior em relação àqueles com sarcoma de tecidos moles em outras localizações.10As vias de disseminação do carcinoma uretral seguem sua subdivisão anatômica. A uretra anterior possui drenagem linfática para região inguinal superficial e profunda. A uretra posterior drena para os gânglios linfáticos da artéria ilíaca externa e hipogástrica, e do músculo obturador interno.10Em um terço dos pacientes, o diagnóstico é tardio, com metástases para gânglios linfáticos inguinais, e em 20% para os gânglios pélvicos. Os locais de metástases a distância mais comum são pulmão, fígado e ossos.10Os tumores anteriores são de melhor prognóstico, chegando a 60% em 5 anos. Os tumores <2cm têm sobrevida de 81% em 5 anos. Em contrapartida, tumores da uretra posterior possuem taxa de 10% de sobrevida em 5 anos se <2cm, 37% de sobrevida em 5 anos quando de 2 a 4cm de tamanho e apenas 7% quando >4cm. Acima do estádio T2, costumam apresentar uma taxa de sobrevida <26% em 5 anos.10As características histológicas dos sarcomas urinários são bastante variadas. A maioria contém alto grau de diferenciação com focos isolados de metaplasia escamosa.7O segundo foi de um homem de 65 anos com uretrostomia perineal para tratamento de estenose uretral distal8, com margem cirúrgica positiva em um paciente com miocardiopatia clinicamente grave, resultando em desfecho desfavorável. O IAM apresentado pelo paciente no período pós-operatório precoce inviabilizou o tratamento adjuvante com radioterapia e quimioterapia. Fica evidente que esses tumores são extremamente agressivos e de mau prognóstico.


Stage 4S bilateral adrenal neuroblastoma presenting in the neonatal period is extremely rare. A 1-day-old male with 4Sbilateral adrenal neuroblastoma complicated by marked hepatomegaly managed by chemotherapy is being reported. The provisional diagnosis of neuroblastoma was made in the fetal life during the last trimester of pregnancy. Cardiomyopathy due to doxorubicin cytotoxicity developed over ensuing years, which is being treated. Stage 4S neuroblastoma (NB) represents approximately 7–10% of all NB cases.[1] Stage 4S NB is now defined by the International Neuroblastoma Staging System (INSS) as primitive tumor (stage 1 or 2) with dissemination restricted to the liver, skin, and bone marrow(less than10%),in children younger than 1 year of age at diagnosis. Bilateral synchronous, multifocal tumors have been included in the definition.[2] According to the International Neuroblastoma Risk Group(INRG) Staging System, the age is extended to 18 months for the stage 4S tumor which is now called stage MS. MIBG scintigraphy must be negative in bone and bone marrow.[3] Stage 4S NB is a metastatic disease associated with good survival, despite a large tumor burden, as spontaneous regression frequently occurs. It has the highest rate of spontaneous regression among malignant tumors. However, in some infants, particularly less than 2 months of age rapid disease progression can be observed with severe functional impairment, with an estimated mortality rate of 10% to 20%.Bilateral adrenal NB is rare.[1-3] The literature search revealed60 cases of bilateral adrenal neuroblastoma. We report a 1-day-old male with 4S bilateral adrenal neuroblastoma complicated by marked hepatomegaly that was managed by chemotherapy.A 1-day-old male neonate with birth weight of 2.5kg was admitted with marked abdominal distension noted at birth. The baby was active with stable general condition. Antenatal ultrasonography report revealed the diagnosis of neuroblastoma during the last trimester of pregnancy. On examination there was no jaundice or subcutaneous nodules. Upper abdomen was distended and revealed a firm, markedly enlarged liver with smooth surface and sharp margins, moving with respiration and reaching the lower abdomen. A distinct large abdominal mass in the left flank was also palpable. Complete blood counts, liver and renal function tests were normal. Serum vanillylmandelic acid (VMA) was 12 mg/24 hours. LDH-2692 U/L and serum Ferritin-400 ng/ml. Radiographs showed calcification in the left upper abdomen along with enlarged liver silhouette. Ultrasonography revealed hepatomegaly and bilaterally enlarged solid cystic masses in suprarenal areas, displacing the kidneys inferiorly. Contrast-enhanced computed tomography (CECT) scan of chest, abdomen and pelvis showed heterogeneous masses in both adrenal glands, larger on left side showing solid cystic areas and calcifications, measuring 60 mm x 48 mm. Right side lesion measured 31 mm x 23 mm . Liver was diffusely enlarged with numerous hypodense lesions in both lobes suggestive of metastasis . There was no retroperitoneal lymphadenopathy, pleural effusion, or bone marrow involvement.Exploratory laparotomy was performed for surgical staging and tissue diagnosis. There was no ascites. A purple, enlarged liver with diffuse solid and cystic nodules on the surface were detected along with bilaterally enlarged adrenal glands. A biopsy was taken from the left adrenal for confirmation of the pathology. Hematoxylin and eosin (HE) staining revealed abundant clusters of small, round, blue cells with high nucleus-to-cytoplasm ratio and immature chromatin. Immunohistochemical (IHC) analysis was positive for chromogranin A (CgA) and synaptophysin. The final diagnosis was bilateral adrenal neuroblastoma, stage 4S with metastasis to the liver.Patient was managed by adjuvant chemotherapy with 4 drugs, cyclophosphamide, cisplatin, etoposide, and doxorubicin over 8 months. There was complete regression of the liver metastasis along with calcification of the bilateral adrenal tumors. Three-year follow-up indicated neither recurrence nor residual tumors. Cardiomyopathy due to doxorubicin toxicity developed over ensuing years, which is being treated with digoxin, enalpril and frusemide. Child is otherwise active and playful.Treatment of 4S NB, particularly those younger than 2 months, with massive hepatomegaly, organ compromise and risk of death, is low-dose to moderate-dose chemotherapy with or without liver radiotherapy.[6] These patients benefit from prompt initiation of therapy, to push the NB cells towards the regression pathway. If this is achieved early, further intensive treatment may possibly be avoided.[1] It is difficult to identify infants with stage 4S disease, who will benefit from chemotherapy.[4] Those with favorable prognostic parameters should be kept under strict observation with supportive care for spontaneous regression to occur.[6]Resection of primary tumor is not associated with improved outcome.[6] A 4S NB complicated by massive hepatomegaly can be managed by abdominal decompression surgery. We operated child for diagnostic purpose as skin lesions and bone marrow involvement were not evident. Low dose radiotherapy to the liver is administered in doses of 100-150 Gy/day. Resolution of liver metastasis is probably related more to the natural course of stage 4S than to radiotherapy.[7] Most common cause of death in stage 4S neuroblastoma is hepatomegaly. Other causes are infection, disseminated intravascular coagulation, and radiation nephritis. Children with 4S neuroblastoma with bilateral adrenal tumors have a good prognosis.To summarize, bilateral adrenal 4S NB may be noted on antenatal ultrasound. High index of suspicion is required in cases where isolated hepatic involvement is found. Biopsy may be required to make a definitive tissue diagnosis. Treatment with chemotherapy is sufficient to control such tumors.Source of Support: NilConflict of Interest: None declared


A 71-year-old man with osteoarthritis and chondrocalcinosis came to our observation developing a swelling in the groin region after a recent left colectomy for adenocarcinoma. The imaging techniques revealed the presence of an iliopsoas bursitis in connection with the hip. The synovial fluid analysis detected the presence of calcium pyrophosphate (CPP) crystals and allowed the final and unusual diagnosis of iliopsoas bursitis related to acute CPP crystal hip arthritis. Hip pain sometimes could represent a challenge even for expert clinicians and could require many imaging techniques efforts to complete the differential diagnostic workup. The hip is one of the most complex joints of the human body, surrounded by a significant number of ligaments and bursae that complicate the detection of the origin of a clinical problem. The iliopsoas bursa  is one of the largest articular recesses of the human body . It liesA 71-year-old Caucasian man, with a history of osteoarthritis and knee chondrocalcinosis , never cThe physical examination revealed the swelling area that could easily be appreciated around and directly above the hip. The hip range of the movement was extremely limited. The patient was able to walk without crutches, keeping his right hip flexed and avoiding putting weight on it. The patellar and achilles reflexes were normal while the strength of quadriceps was decreased.The laboratoristic parameters showed an augmented C-reactive protein , while erythrocyte sedimentation rate, anticitrulline antibodies, and rheumatoid factor level were normal as the screening for reactive arthritis.Magnetic resonance imaging (MRI) of the pelvis revealed an elongated neoformation with predominantly liquid content, multiloculated and localized in the anterior-lower aspect of the right hip joint, extending longitudinally for 6.5 cm and in diameter axial for 3.1 cm with multiple partitions in the context . The MRI18F-FDG PET). In the fusion images (PET/CT), a moderate uptake of the radiopharmaceutical drug (18-fluorodeoxyglucose) in the anterior and inferior part of coxofemoral right is appreciated, more evident in the peripheral portion, and intense storage of the radioisotope in the hip joint intra-articular level  and a fluorine-18-fluorodeoxyglucose positron emission tomography , equipped with linear probe with broad frequency band (6–18 MHz) and the Doppler frequency of more than 9 MHz. Axial and sagittal images were obtained while the patient was in a supine position, putting the probe over the hip. In particular, the longitudinal scan demonstrated the presence of a predominantly liquid content before the right coxofemoral joint, associated with synovitis of the hip. Semicoronal scanning, instead, showed the presence of the connection between the hip joint and the neoformation .3, normal value <200; proteins: 5.16 g/dL, normal value 1–2.5) was aspirated from the bursa. Surprisingly, the optical microscopy fluid analysis revealed the abundant presence of small rhomboid crystals with a weak positive birefringence.A turbid fluid with diminished viscosity and inflammatory features (leukocytes count: 8600/mmThe following cytological examination was negative for neoplastic cells and the bacteriological examinations did not detect any pathogen agent.The final diagnosis was iliopsoas bursitis related to acute CPP crystal hip arthritis.Colchicine in association with an oral steroid course determined a prompt remission of the articular and bursal inflammatory signs.Iliopsoas bursitis is usually due to an excessive production of synovial fluid in the hip. While the pathogenesis is not well known yet, it is postulated that the collection of fluid in the bursa is associated with a high pressure effusion inside the joint. Then, the fluid is pushed from the joint to the bursa, a zone with a lower pressure . In thisFor other authors, the bursitis is a simple protrusion of the joint capsule weakened by degenerative changes . Indeed,Rheumatoid arthritis frequently underlies an enlargement of iliopsoas bursa –5. In thIn our case, we believe that the synovial fluid originated from the joint, considering the detection of CCP crystals inside the bursal fluid and the calcifications of the cartilage of the acetabular head. The inflammatory action of CCP crystals induced an acute production of synovial fluid and, then, the increased pressure inside the hip allowed the bursal collection. The inflammation induced by CCP crystals could be so powerful to produce a complete hip destruction within few weeks . Even ifIn regard of the recent colic neoplasm, an intensive and complete diagnostic route was performed, starting from the imaging techniques. The other possibilities of this kind of swelling in the groin region considered were a metastasis, a lymphocele, a partially colliquative lymph node, or a hematoma.Finally, the synovial fluid analysis avoided a new surgical intervention to resect the cyst, and the final diagnosis was established accordingly to the European League against Rheumatism recommendations for calcium pyrophosphate deposition .In the evaluation of a patient with pain and/or swelling in the hip region and with symptoms related to the lower limb difficult to explain, the presence of an iliopsoas bursitis should be kept in mind.


The differential diagnosis of a mediastinal mass is a common challenge in clinical practice, with a wide range of differential diagnosis to be considered. One of the rarer causes is tuberculosis. Atypical presentations of tuberculosis are well documented in immunocompromised patients, but should also be considered in the immunocompetent.This case outlines a previously healthy 22 year-old immunocompetent male presenting with worsening chest pain, positional dyspnea, dry cough and dysphagia. Chest x-ray showed evidence of an isolated anterior mediastinal mass, which was confirmed on computed tomography. A mediastinoscopy was diagnostic as histology revealed necrotizing granulomatous inflammation and the presence of acid-fast bacilli, indicating mediastinal tuberculosis.Typically the underlying presentation of mediastinal tuberculosis is mediastinal lymphadenitis. This case was unusual in that we detected an isolated large anterior mediastinal mass accompanied by a relatively small burden of mediastinal lymphadenitis. Cases similar to this have been documented in immunosuppressed patients however in our case no evidence of immunosuppression was found. This case report emphasizes the importance that a detailed and logical pathway of investigation is pursued when encountering a mediastinal mass.The online version of this article (doi:10.1186/s12890-016-0175-7) contains supplementary material, which is available to authorized users. Mycobacterium tuberculosis undergoes lympho-heamatogenous dissemination after entering the respiratory tract. Typically the mediastinal and hilar lymph nodes are the first lymphatic tissues M. tuberculosis will encounter . Pati. Pati4].There are several and varied presentations of mediastinal tuberculosis including lymph node calcification, oesophagomediastinal fistula, pericardial TB and fibrosing mediastinitis. The underlying cause of these lesions is mediastinal lymphadenitis, which extends to involve surrounding structures creating strictures and fistulation . This caThere are few documented cases of tuberculosis presenting as an isolated anterior mediastinal mass in an immunocompetent patient. Comprehensive review of the literature reveals ten similarly described cases with the majority occurring in the pediatric population aged 2 years or younger. Of the published cases on adults all three patients were male with age ranging from 32 to 76 years –14. The In summary, while the aetiology of a mediastinal mass can be difficult to establish because of the wide differential, it is imperative that a through and logical scheme of investigation including tissue diagnosis is pursued. This case aims to highlight the importance of considering tuberculosis in the differential even in immunocompetent patients presenting with an isolated mediastinal mass that cannot otherwise be explained.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor of this journal. This project was carried out within an appropriate ethical framework. This manuscript conforms with the CARE guidelines published in 2013 (see Additional file Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor of this journal.N/A.


Both mucinous cystadenoma of the appendix and intestinal schistosomiasis are rare lesions. We report a rare case of simultaneous giant mucinous cystadenoma of the appendix and intestinal schistosomiasis. A 64-year-old man from China presented with a one-year history of pain in the right lower quadrant of the abdomen. There were no other pertinent historical findings, other than schistosomiasis. Imaging showed a large, tubular, mesenteric cystic structure extending downwards from the inferior wall of the cecum. Right hemicolectomy was performed for the appendiceal tumor. The final pathological diagnosis was mucinous cystadenoma with calcified Schistosome eggs within the mucosa and submucosa of the appendix, small intestine, colon, and lymph nodes. We deduced that the pathogenesis of appendiceal mucinous cystadenoma in our case was Schistosome eggs causing luminal obstruction, finally resulting in intraluminal accumulation of mucoid material. Postoperatively, the patient recovered well. A mucocele is an uncommon pathology of the appendix, characterized by an obstructive dilatation of the appendix caused by intraluminal accumulation of mucoid material. The incidence is observed in 0.2% to 0.7% of all appendectomied specimens , 2. AccoThe patient was a 64-year-old man who was born in Yueyang City, Hunan Province, in Southern China, one of the most heavily endemic areas with schistosomiasis over the past five decades. The patient was admitted to our department with a one-year history of intermittent pain in the lower right quadrant of the abdomen. When the lower right quadrant of the abdomen was palpated, the patient felt pain, but his muscles were not rigid and there was no rebound tenderness. On physical examination, there was no palpable mass in the right lower abdomen. No liver or spleen enlargement was noted. Abdominal ultrasonography revealed a well-encapsulated hypoechoic process measuring 103 × 43 mm Figure . At the A mucocele of the appendix is a descriptive term that refers to dilation of a lumen because of accumulation of a large amount of mucin. It is a rare lesion in all resected appendiceal tumors. Schistosomiasis of the appendix is also a rare lesion and is observed in 0.4% of all appendectomies performed . SimultaThe characteristic pathological tissue response of appendiceal schistosomiasis is believed to be a granulomatous inflammatory reaction to the schistosomal ova. There is formation of epithelioid cell granulomas, predominantly in the submucosa and serosa, which ultimately undergo fibrosis. The final pathologic examination of the present case revealed calcified Schistosome eggs within the mucosa, submucosa of the appendix, small intestine, colon, and lymph nodes. Considering that mucoceles are caused by mucus distention secondary to an obstruction of the appendix, we speculate that the pathogenesis of appendiceal mucinous cystadenoma in our case is granulomatous inflammation and fibrosis reaction to the Schistosome eggs causing luminal obstruction, finally resulting in intraluminal accumulation of mucoid material.Mucoceles may be caused by one of four processes: cyst retention, mucosal hyperplasia, mucinous cystadenoma, or mucinous cystadenocarcinoma. Our patient belongs to the mucinous cystadenoma group. Mucinous cystadenoma is a benign tumor characterized by marked distention of the lumen, of up to 6 cm.et al.[The clinical presentation of appendiceal mucoceles is not specific. Very large lesions (up to 51%) will be asymptomatic . A studyet al. showed tPreoperative diagnosis is difficult but is very important for the selection of an adequate surgical method. Computed tomography, ultrasonography, and colonoscopy are useful for preoperative diagnostics. Computed tomography is the most accurate method in the diagnosis of a mucocele: a typical finding of an appendiceal mucocele is a well-encapsulated cystic mass 2 to 20 cm in diameter communicating with the cecum. Ultrasonography can be used to differentiate between a mucocele and acute appendicitis. For patients with acute abdominal pain in the right lower quadrant of the abdomen, ultrasonography is the first-line diagnostic method. For mucoceles, ultrasonography reveals a sausage-shaped cystic structure in the region of the appendix. Colonoscopy may show elevation of the appendiceal orifice. In some cases, colonoscopy may show the ‘volcano sign’; the appearance of the appendiceal orifice at the center of the mound . ColonosTreatment for appendiceal mucoceles mainly relies on surgery and partly requires chemotherapeutic washing of the abdomen . One of We report a rare case of simultaneous giant mucinous cystadenoma of the appendix and intestinal schistosomiasis and provide a brief review of the literature.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editor-in-chief of this journal.


An 73-year-old man, with no abdominal surgical history, presented to the emergency in an array of acute intestinal obstruction. Abdominal CT-Scan axial (A) and coronal (B) sections were performed objectifying hernia sac containing intestinal structure hail externalized through a collar on the outer edge of the rectus muscle on Spiegel's line with no sign of digestive sufferance. The laparoscopic total extraperitoneal mesh repair of the Spiegel hernia was performed. The postoperative course was uneventful and the patient left the hospital on the seventh postoperative day after resumption of normal intestinal transit. Spiegelian hernia is a rare type of ventral abdominal hernia. It occurred in patients in the 6th and 7th decade of life, with an equal sex prevalence. The pathogenesis often involves a dehiscence of the transverse and internal oblique muscle aponevrosis. The diagnosis of Spiegelian hernia is difficult because no characteristic symptoms are identified and often there is no palpable mass, therefore only 50% of cases are diagnosed preoperatively. The abdominal CT scan is helpful in the description of hernia's topography and sometimes in diagnostic confirmation. Surgical treatment is recommended due to the high risk of complications like incarceration and strangulation.


Sclerosing xanthofibroma is a benign lesion generally of flat bones that is thought to be caused by a reactive response to intramedullary hemorrhage following chest wall trauma. We are reporting a case of a 56-year-old man that was complaining of a dump pain on the right back and a swelling right in this place for several weeks. The radiology was suggesting an aggressive malignant tumor of the chest wall and probably metastasis in both lungs meanwhile the patient was in good state and very active. The surgery was decisive for the diagnosis that, to the fortune of the patient, it was sclerosing xanthofibroma. The tumors of thoracic wall are numerous and vary from benign lesions to very aggressive malignant ones with a poor prognosis. In most of them the radiology is very helpful for the diagnosis, but the biopsy is the most decisive diagnostic procedure. One of the benign lesions of the thoracic wall that is rarely found is sclerosing xanthofibroma of the rib –7.This is the case of a 56-year-old man that was complaining of a dump pain on the right back and a swelling right in this place for several weeks. The patient was in good state and very active. There was not any health problem in the past except a thoracic trauma at work one year ago. In that time the patient was diagnosed with a simple fracture of the 9th right rib without any other consequences.On the X-ray was seen a shadow in the lower part of the right hemithorax. After that, it was decided to perform a CT-scan of the thorax that revealed a tumor of the thoracic wall in the right hemithorax that measured 8 × 4 cm and had a heterogeneous density inside of it. The tumor had involved and destructed the 9th rib and was lying even in two adjacent intercostal spaces, but without involving the lung and muscular layers. On lung window of the CT-scan were seen micronodular infiltrations of both lungs with diameters up to 5 mm and only one nodule in lower lobe of the right lung with diameter almost 1 cm . The medBecause there was not a correlation between the clinical picture of the patient and the conclusion of the radiology it was decided to perform frozen biopsy of the lesions of the right lung and an excision biopsy of the tumor of the thoracic wall. So, through a small posterior thoracotomy at 7th intercostal space were sampled five nodular lesions from the right lung and a sample from the tumor. None of these samples resulted positive  after frozen biopsy. In such conditions was considered as more realistic the option of performing an oncologic resection of the tumor of the thoracic wall.  After that, we have performed a plastic procedure with polypropylene mesh in double layers to correct the defect. The clinical course of the patient was very good and five days later he was discharged from the hospital in very good condition. The conclusion of biopsy for both the tumor of thoracic wall and the lung nodules was sclerosing xanthofibroma which is considered by most people to be a tumor with different clinical and radiological features. Lesions were characterized by a network of anastomosing bone trabeculae without osteoblast lining within a fibrous stroma .Referring to the follow-up, the patient was in a great state of health and from the first month after the surgery he turned back at his work place. Two years after the surgery the thoracic CT-scan showed neither recurrences of the lesions of the thoracic wall nor new developments on the lungs.The exact nature of sclerosing xanthofibroma remains controversial. Most of authors have considered being developmental lesions rather than true neoplasms. For some authors it is considered as a benign variant of malignant fibrous histiocytoma. The lesion is believed to be a reactive response to intramedullary hemorrhage following chest wall trauma. Almost all the reported cases of sclerosing xanthofibroma have occurred in patients older than 20 years of age and most of them were accompanied by pain. The lesions in sclerosing xanthofibroma are described as lytic, most with sharply defined margins and a sclerotic rim. The lesions are small and most commonly involve the flat bones, especially the pelvis or ribs. In long bones, the metaphysis, epiphysis, or diaphysis may be involved. In 70 cases of sclerosing xanthofibroma known and reported in a study of Blanco et al. only six of them belong to the rib . CorticaThe differential diagnosis of sclerosing xanthofibroma has to be done with nonossifying fibroma and fibrous cortical defect that are not true neoplasms, only developmental defects. The differential diagnosis has to be done even with giant cell tumor, giant cell reparative granuloma, eosinophilic granuloma, osteosarcoma, and so forth –7.Sclerosing xanthofibroma of the rib is a rare tumor of the chest wall that radiologically may resemble very much a malign tumor but does not behave as it.The clinical status of the patient that does not show such a great sufferance having a suspected malign tumor and the history of a trauma of the thoracic wall must not be ignored. However the clue of the diagnosis for sclerosing xanthofibroma is the biopsy.Although according to the literature the recurrences do not happen after a partial resection, we believe that when the diagnosis is not sure and rare as in our case, at least the resection of the interested rib is necessary for a clear diagnosis and for the releasing of the pain.


There are a lot of different causes of abdominal pain; in this case, a young woman suffers from three diseases with similar symptoms. Adult intestinal mal-rotation is a rare condition of deviation from the normal 270° counter clockwise rotation of the midgut resulting in, not only mal-position of the small intestine, but also mal-fixation of the mesentery. Fitz-Hugh-Curtis syndrome is a rare complication of pelvic inflammatory disease; it involves liver capsule inflammation associated with genital tract infection, which is usually caused by Neisseria gonorrhoea and Chlamydia trachomatis. Neuroendocrine tumors are enterochromaffin cell neoplasms that arise from cells of the endocrine  and nervous systems; the appendicular one is the most common primary malignant lesion of these tumors, it’s incidence is about 0.3 – 0.9 % of appendectomies done. Just for knowledge, this is the first described case of concomitant presence of all these diseases with clinical symptoms attributable to each one.40-years-old woman suffers from acute abdominal pain, predominantly on the right quadrants, without abdominal distension, no guarding nor rigidity and normal intestinal peristalsis. She has a long history of abdominal intermittent pain, with cramps every 30–40 min, resolving spontaneously. She was diagnosed as intestinal mal-rotation through computed tomography scan which has evidenced a mobilized intra - peritoneal duodenum with cecum/ascending colon predominately lying on the left side and the small intestine almost entirely lying on the right side of abdomen, without evidence of effusion, edema or signs of intestinal ischemia or infarction. Exploratory laparoscopy demonstrated an inflammatory process in the hepatic-renal space, with bloody adhesions above the liver capsule; this is additional to the typical pelvic inflammatory disease signs (Fitz-Hugh-Curtis syndrome). Appendectomy was performed with histological analysis resulting in appendicular neuroendocrine tumor.Although the patient has an intestinal mal-rotation which could explain the abdominal painful symptoms, it is not possible to exclude other concomitant causes, such as perihepatitis on pelvic inflammatory disease or neuroendocrine tumors. Even if all these diseases are rarely seen in daily clinical practice, they should be considered in the differential diagnosis of chronic intermittent abdominal pain in a young woman. Intestinal Mal-rotation (IM) is defined as any deviation from the normal 270° counter clockwise rotation of the midgut during embryological development, resulting not only in the mal-position of the small intestine but also in the mal-fixation of the mesentery . It is rFitz-Hugh-Curtis syndrome is a complication of pelvic inflammatory disease (PID); it involves liver capsule inflammation associated with genital tract infection, which is usually caused by Neisseria gonorrhoea and Chlamydia trachomatis. The main symptoms are acute abdominal pain, usually in right upper quadrant, sometimes associated also with signs of salpingitis; chills, nausea, vomiting, hiccupping and malaise are commonly reported. The incidence ranges from 4 to 14 % in female with PID, reaching 27 % in adolescents cases.Neuroendocrine tumors (NETs) are enterochromaffin /neuroendocrine cell neoplasms with special secretory granules capacity that may determine a carcinoid syndrome, including symptoms as abdominal pain, diarrea/constipation, nausea/vomiting, jaundice or changing in stool colour. NETs most commonly occur in the gastrointestinal tract (67 %) and bronchopulmonary system (25 %). The appendicular one is the most common primary malignant lesion originating in the appendix, found in 0.3 – 0.9 % of appendectomies done [We report an unusual case of 40-years-old female with a long history of intermittent abdominal pain, especially on the upper right quadrant, already studied by numerous medical centres without certain definitive diagnosis; we detected an intestinal mal-rotation , also we diagnosed a Fitz-Hugh-Curtis syndrome which is usually caused by Chlamydia trachomatis and an appendicular NETs. This rare case indicates that, even if the patient has an intestinal mal-rotation, which could explain the abdominal symptoms, it is not possible to exclude another concomitant causes, as C. trachomnatis infection, PID and NETs.A 40-years-old woman presented to the Emergency Department with acute intermittent abdominal pain with cramps, located in all right quadrants, started from 24 to 48 h. The physical examination results in palpable abdomen without abdominal distension, nor guarding or rigidity and normal intestinal peristalsis; the liver and spleen were not palpable. The clinical evaluation showed absence of fever, nausea, vomiting or modifications of the bowel habit, absence of any abnormal urological symptoms and normal vital signs. All the blood and urine analysis were normal. The history of the patient is free of any abdominal surgical operations. About the sexual history, she was nulliparous, free of dyspareunia, pelvic pain, spotting, itching or other gynaecological symptoms. The patient had in the past some similar episodes of abdominal intermittent pain associated with cramps recurring every 30–40 min, resolving spontaneously, usually two times/year during the past three years; an ultrasound, computed tomography (CT) scan and upper/lower endoscopy have evidenced an intestinal mal-rotation. Now we have repeated the CT scan which has evidenced a mobilized intra-peritoneal duodenum Fig.  in all oth week to the 10th week of embryonic development. The midgut lengthens disproportionately during this period and undergoes rotation around the SMA axis for a total of 270° in the counter clockwise direction. Secondly (Stage II), the midgut loop comes back into the abdomen, approximately from the 10th week to the 11th week; when re-entered into the abdominal cavity, the cephalic midgut completes its 270° counter clockwise rotation while the caudal midgut also completes its rotation, resulting in the duodenum coursing inferior and posterior to the SMA and the caecum located in the right lower quadrant. Stage III lasts from the end of Stage II until just after birth, completing the fixation. The descending and ascending colon mesenteries fuse with the retro peritoneum, the small bowel is fixed by a broad mesentery from the duodenojejunal junction in the left upper quadrant to the ileocecal valve in the right lower abdomen. One of the most frequent forms is “non-rotation”, where the first and second parts of the duodenum are situated normally but the third and fourth parts descending vertically downward along the right side of the superior mesenteric artery, the small bowel on the right and the colon doubled on itself to the left of midline; in “reversed rotation” there is the caecum and colon positioned posterior to the superior mesenteric vessels, the duodenum subsequently crosses anterior to it; finally, “mal-rotation” is a spectrum of abnormalities, the most frequent of which are duodenojejunal flexure located inferiorly in the right of the midline and caecum in a sub hepatic or central position [IM is a congenital anomaly, usually diagnosed in the first month of life (nearly 85 % of cases), while it’s rarely found in adulthood about 0.2–0.5 %); it has been estimated to occur in approximately one in 500 live births, but the true incidence in old age is unknown because the major part of patients are asymptomatic –0.5 %; i. The intposition . If the position ; but in position . In 1978position  demonstrposition . Accordiposition . The incposition . The etiposition . Diagnosposition . The mosposition . The thememento; often the diagnosis is not simple.In our case, in the context of a generalized pelvic inflammation, the appendix appeared moderately inflamed, so we performed a laparoscopic appendectomy; the histological report was of appendicular NET (pT1a G1). The neuroendocrine tumors are so called for the enterochromaffin/neuroendocrine cell origin with neurosecretory capacity, producing and secreting different hormones; many of these products may rise to hormonal syndromes, especially the insulinomas, glucagonomas, gastrinomas and serotoninomas. The appendicular NETs are the most common appendicular tumor, mainly frequent in women  of 40–50 years old . AppendiWritten informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.


Desmoid tumor is a rare, benign, usually asymptomatic fibromatous lesion. The etiology is unknown and the diagnosis is based on histopathological examination. The treatment is complete resection of the tumor. Pancreatic desmoid tumor is extremely rare. In the literature there have been only 11 cases described, most of them as solid or solid-cystic masses. We report the case of a patient with an isolated cystic pancreatic desmoid tumor that is, to the best of our knowledge, the second reported case.en bloc resection of the left colonic flexure was performed. Histological analysis confirmed that the resection was complete. The mass had infiltrated the pancreatic parenchyma. All tumor cells were positive for anti-beta-catenin staining characteristic for desmoid tumor. No abnormalities in the spleen and colon were found.A 13-year old Caucasian boy presented with recurrent pain of two months’ duration in the left hypochondrium of his abdomen. An ultrasound examination and computed tomography scan revealed the presence of a cystic mass located in his splenic hilum, tightly adjacent to the pancreatic tail. A splenic cyst was suspected. Operative findings showed a 10x10cm cystic mass tightly connected to the pancreatic tail and left colonic flexure, adherent to the spleen, splenic vein and artery. Distal splenopancreatectomy with Isolated sporadic pancreatic desmoid tumor with cyst formation is extremely rare and its diagnosis can be difficult, especially because of uncharacteristic symptoms and radiological findings, as in our patient. This case report should be of interest not only to surgeons, as the treatment of choice is radical resection, but also gastroenterologists, considering it is in close relation with familial adenomatous polyposis, and oncologists as the reason for differentiation with other pancreatic tumors. APC gene mutation) [Desmoid tumor (DT) is a particular type of fibromatosis. It is a rare fibromatous lesion that is a result of abnormal proliferation of myofibroblasts. DT generally involves fascial and musculoaponeurotic tissues. It comprises 0.03% of all tumors and 3% of fibrous tissue tumors ,2. We cautation) ,3,6. Theutation) .Isolated pancreatic DT is extremely rare. In the literature there have been only 11 cases described of pancreatic DT with eight located in the tail. Seven out of 11 published cases were solid mass, with only three solid-cystic and one cystic mass ,4,6,7.We report the case of a patient with an isolated cystic mass located in the pancreatic tail diagnosed by beta-catenin immunohistochemical staining as a DT; it is, to the best of our knowledge, the second reported case.Echinococcus antibodies were negative. Our patient was qualified for radical surgery. Before the operation, the patient was vaccinated with pneumococcal and meningococcal vaccines as recommended in cases of elective splenectomy. Operative findings showed a 10x10cm cystic mass with a thick, grey white wall, tightly connected and macroscopically invading his pancreatic tail and left colonic flexure, spleen, splenic vein and artery, but not his stomach and left kidney  scan revealed the presence of a cystic mass (10x7x9cm) with multiple septums located in his splenic hilum, tightly adjacent to the pancreatic tail and the left kidney  are normal and show no recurrence of the DT.en bloc resection of the left colonic flexure with histopathologically confirmed uninvolved margins was the reason for no complementary therapy. Our patient remains disease-free 19 months after the surgery. Taking into account the close relationship between DTs and FAP, investigations for colonic changes should be undertaken. In our case, colonoscopy revealed no changes in the colonic wall.Desmoid tumor is a very rare fibromatous lesion, a result of abnormal myofibroblast proliferation. DTs are benign, do not metastasize but can be locally aggressive. Localization of DTs can be abdominal or pelvic, extra-abdominal or intra-abdominal. Intra-abdominal DTs are the rarest and mostly associated with FAP. Pancreatic DTs, as described in our patient, are extremely rare, are mostly located in the tail of the pancreas, and occur as a solid mass. To the best of our knowledge, only three cases from those already reported were with a cystic component and only one case occurred as a cyst like in our patient. DTs are usually asymptomatic for a long period of time or can cause uncharacteristic pain as observed in our case. An initial diagnosis of DT based on radiological findings and laboratory tests is impossible. It requires differentiation from other abdominal cystic tumors. The final diagnosis can be based only on histological and immunohistochemical findings . In our Isolated sporadic pancreatic desmoid tumor with cyst formation is extremely rare and its diagnosis can be difficult, such as in our case, where it preoperatively presented as a splenic cyst. Even though DTs are benign and do not metastasize, they can be locally aggressive. It is considered that when possible, the treatment of choice for DT is radical resection with clear margins, as in our patient.Written informed consent was obtained from the patient’s legal guardian(s) for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Intracholecystic papillary-tubular neoplasms are rare precursor lesions of gallbladder cancer. They were proposed as a separate pathologic entity in 2012 by Adsay et al. for the unification of a variety of mass-forming precursor lesions including papillary adenomas, tubulopapillary adenomas, intestinal adenomas, and others. They are considered homologous to intrapapillary mucinous neoplasms of the pancreas and intrabiliary papillary neoplasms of the common bile duct. In contrast with the commoner flat-type precursor gallbladder cancer lesions, they follow a more indolent clinical course and probably different genetic pathways to carcinogenesis. They are largely uninvestigated with only a handful of studies providing biological and clinical information. Choledochal cysts are dilation of the common bile duct. Diagnosis is usually established during childhood, and only a minority of patients are diagnosed at adulthood. They are of major clinical importance as they are known predisposing factors for biliary carcinogenesis.The current report describes a patient with a simultaneous diagnosis of choledochal cyst and intracholecystic papillary-tubular neoplasm. The patient underwent excision of the extrahepatic biliary tree for a Todani I choledochal cyst, and histological examination of the specimen revealed an intracholecystic papillary-tubular neoplasm of the gallbladder. Authors describe diagnostic and clinical course of the patient alongside clinical and biological characteristics of these rare lesions.To the best of our knowledge, this is the first report of a patient with a simultaneous diagnosis of choledochal cyst and intracholecystic papillary-tubular neoplasm. Those rare lesions shed light on different forms of gallbladder cancer carcinogenesis and its relationship with choledochal cysts and cholestasis. Precursor lesions of gallbladder cancer separate into two categories: those exhibiting flat dysplasia and the mass-forming lesions. The former are commoner and proceed to carcinogenesis through a metaplasia-dysplasia cancer pathway , 2. The ICPNs are rare neoplasms of the gallbladder, homologous to pancreatic IPMNs, and the recently described IPBNs. Adsay et al.  define tSince their first description in 2012 by Asday et al. , only twICPNs are considered analogous to IPBNs and to a lesser extent to IPMNs. This is supported by their exophytic nature, expression of the same cellular lineages, similar clinical behavior, and better survival. Furthermore, ICPNs seem to progress to cancer through an adenoma-carcinoma sequence rather than the much commoner metaplasia-dysplasia-carcinoma sequence , 8. UntiMacroscopically, they are polypoid lesion presenting large villous/papillary growths with smooth surface projections. They can be sessile or pedunculated . AccordiA more benign clinical behavior than flat type precursor lesions characterizes ICPNs. ICPNs are incidentally found at only 0.4 % of cholecystectomies. Approximately 6.4 % of gallbladder cancers carry an ICPN component, indicating that ICPNs can progress to gallbladder cancer. Stage-matched comparison between typical gallbladder cancer and invasive ICPNs proved that ICPNs metastasize less often to lymph nodes and have better disease-free and overall survival , 8, 13. Choledochal cysts are rare congenital dilatations of the biliary track. Although benign, their presence is associated with serious complications including biliary carcinoma, pancreatitis, and choledocholithiasis . Their iCholedochal cysts are predominantly present in children. They are four times commoner in females. In developed countries, only 20–30 % of the patients are diagnosed at adulthood . PatientEtiology of choledochal cysts is unknown. Their pathophysiology is closely related to the existence of pancreatic maljunction, i.e., abnormal junction of the common biliary duct and pancreatic duct outside the duodenal wall. It is a rare disorder, occurring in less than 2 % of the population while >80 % of all pediatric choledochal cysts are associated with pancreatobiliary maljunction , 23. SphA young Caucasian female patient aged 28 years came to our hospital complaining of chronic right upper quadrant pain. Physical examination was unremarkable as was her medical history. She was investigated with upper abdominal ultrasound that was negative for chololithiasis but revealed dilated gallbladder and common bile duct with a diameter of 2.1 cm. Her laboratory values were within normal limits with total bilirubin of 0.28 mg/dl, alkaline phosphatase (ALP) at 42 U/l, and γ- GT 16 U/l. Serum tumor markers were within normal limits. The patient was further studied with computerized tomography, magnetic resonance imaging, and magnetic resonance cholangiopancreatography (Fig. With a diagnosis of a choledochal cyst, Type Ia according to Todani classification, the patient underwent excision of the extrahepatic biliary tree with Roux-en-Y reconstruction. An intraoperative cholangiography was performed for diagnosis confirmation Fig. .Fig. 2InThe patient had an uneventful recovery and left the hospital at the seventh postoperative day. Specimen was sent for histological examination Fig. . On histIn this report, authors describe a rare case of ICPN incidentally discovered after excision of the extrahepatic biliary tree for a type I choledochal cyst in a young female patient. We also perform a brief literature review on ICPNs and mechanisms of gallbladder carcinogenesis. To the best of our knowledge, this is the first time an ICPN was reported in a patient with a choledochal cyst.Resende et al.  reportedThose rare precancerous lessions give insight to carcinogenesis of biliary track cancer. Their rarity does not allow concrete conclusion formulation; still, few accumulated evidence indicates a relationship between extrahepatic obstruction and biliary stasis and carcinogenesis in the biliary track. Biliary stasis causes continuous inflammation and epithelium damage, leading the biliary epithelium to regenerative cycles. Inevitable acceleration of the cell cycle makes it susceptible to metaplasia and consequent dysplasia. It seems that this pathophysiological mechanism also causes ICPNs, yet why some patients will follow the more malignant metaplasia-dysplasia-carcinoma pathway and others the more indolent adenoma-carcinoma pathway remains unknown , 27.ALP, alkaline phosphatase; ICPN, intracholecystic papillary-tubular neoplasm; IPBN, intraductal papillary biliary neoplasm; IPMN, intraductal papillary mucinous neoplasm; γ-GT, gamma-glutamyl transferase


Documented reports of gastrointestinal stromal tumors (GIST) are relatively few in the sub-Saharan continent. The body of evidence points towards anal wall involvement being a rarity indeed. In this article we document a 61 year old Nigerian man who presented with bleeding per rectum and in whom the histological features (including immunohistochemistry) of the biopsied anal lesion was GIST. Mazur and Clark were the first to coin the term “Gastrointestinal stromal tumors (GISTs)” in 1983 . The incA 61 year old Nigerian gentleman was referred for colonoscopy in Lagos, Nigeria on account of passage of blood per rectum. The patient had noted the initial episode about 3 months prior to presentation. The blood was noted to precede and also line the periphery of the fecal bulk that was expelled. There was no history of alteration in bowel habit nor did the patient report any other systemic symptoms. The patient was in stable clinical condition on examination and the digital rectal examination had revealed a small, soft mass in the anterior aspect of the anal wall with the withdrawn finger being stained with blood. A packed cell volume test was 41%. Upon retroflexion in the rectum, the colonoscopy findings were that of a 5 cm in diameter polypoidal mass in the anterior wall of the anal canal. The mass had multiple umbilications on its irregular surface and was noted to be gently oozing blood at the time of examination. Cold forceps were used to biopsy the lesion and samples were sent for histology. The clinical suspicion of a malignant tumour was raised. An abdominal computed tomography (CT) scan revealed a small mass in the anal wall with focal mural thickening and minimal fat stranding which is indicative of limited local infiltration. There was no radiographic suggestion of regional lymph node involvement. The microscopic examination showed complete replacement of the tissue by a focally necrotic tumor that comprised proliferating stromal cells with peripheral palisading . NuclearThe above case report details a rare case of GIST of the anal wall in an elderly Nigerian patient. The case, with its full immunohistochemical characterization, to the best of the authors’ knowledge, is the first of such from this region of Africa. The occurrence of anal GIST is very rare indeed as it represents only 3% of ano-rectal mesenchymal tumors . Put in The report documents a rare case of anal GIST as a cause of hematochezia in a patient from sub-Saharan Africa. The need for advanced histopathologic assessment (including detailed immunohistochemitry) of all such lesions to provide further clarity after basic histology stains is further emphasized- even as we note that the resource constrained environment that we live in makes this easier said than done.


Liver abscess formation due to enterohepatic migration of a foreign body is extremely rare. Foreign body ingestion is generally an unconscious and painless event, thus complicating preoperative diagnosis in most patients. We report the case of a 61-year-old man who presented with secondary peritonitis from a ruptured hepatic abscess after an ingested fish bone migrated into the liver.  Enterohepatic migration of an ingested foreign body with liver abscess formation is an extremely rare event. We report the successful management of a patient who presented with secondary peritonitis from a ruptured liver abscess, caused by an ingested fish bone that migrated into the liver.A previously well 61-year-old man presented to our surgical unit with a three-week history of abdominal pain. Clinical examination revealed an anicteric pyrexial patient with generalized peritonitis. A chest roentgenogram showed no free air and a complete blood count and liver function tests were noted to be within normal limits. An abdominal ultrasound was highly suggestive of an abscess measuring 56 mm × 31 mm × 51 mm in the left liver lobe, but there was no radiologically demonstrable foreign body. An exploratory laparotomy was performed which showed free pus in the abdomen; the left lobe of the liver was adherent to the body of the stomach anteriorly. Gentle dissection revealed an abscess cavity in segment three (Couinaud) of the liver containing a fish bone measuring 6 cm in length .Further inspection confirmed no fistulous communication between the stomach and the liver. The abscess was drained, fish bone was removed, and surgery was completed with peritoneal lavage and insertion of a corrugated drain. In the postoperative phase, the patient was managed with parenteral antibiotics and discharged uneventfully.The majority of ingested foreign bodies will pass through the gastrointestinal tract uneventfully. Although the incidence is less than 1%, sharp objects such as toothpicks, sewing needles, and fish bones can result in hollow viscous perforation . The fir


Sertoli-Leydig cell tumor (SLCT) is a rare sex-cord tumor that usually occurs unilaterally and accounts for < 0.5% of all ovarian tumors. SLCT is uncommon in post-menopausal women, with the average age of diagnosis being 25 years.We present a case of a 63-year-old post-menopausal woman presenting with progressive hirsutism, and male-pattern baldness. Unusual nail changes were also observed.Hormonal profile of the patient revealed increased testosterone and estradiol levels, and a 3.5 cm left ovarian mass. The patient was evaluated and was not found to be anemic or iron-deficient. Intraoperative frozen section assessment during laparoscopic exploration revealed SLCT, which was confirmed subsequently by histopathological and immunohistochemical (IHC) examination. Nail bed tissues were collected from normal females and evaluated by IHC for the presence of androgen receptors (AR).The patient had an excellent postoperative course and all her testosterone-related manifestations were reversed within one year of surgery. Following surgery, the patient’s unique nail abnormalities also resolved gradually. The IHC evaluation also confirmed the presence of AR in nail bed tissues of females.SLCT, albeit rare, should be considered in post-menopausal women presenting with virilization and elevated androgen levels. Unusual nail signs may develop in response to increased androgen levels in these patients. Sertoli-Leydig cell tumor (SLCT) is a rare sex cord-stromal tumor that accounts for less than 0.5% of all ovarian neoplasms . OvarianWe present the case of a 63-year-old post-menopausal G3P3003 Caucasian female presenting with complaints of progressively increased hair growth, deepening voice, and a receding hairline over the past year. She also complained of some pelvic discomfort that resembled menstrual cramps, but denied overt vaginal bleeding. Her past medical history was significant for osteoporosis diagnosed twelve years prior. She denied any history of ovarian or adrenal disease. She denied any use of medications. The patient’s age of menarche was at 12 years and menopause at the age of 50 years. The patient’s last Pap smear was a year ago which was normal and she also reported no history of abnormal pap smears. The patient reported three normal pregnancies with uneventful cesarean sections and three living healthy children. Surgical history was remarkable for tonsillectomy and rhinoplasty. She was a happily married homemaker and exercised regularly. Interestingly, she noted an increased exercise tolerance and reported that she could easily double her normal biking routine. She also reported an increased libido. She denied tobacco, alcohol, or intravenous drug use. Family history was significant for hypertension in her mother and cardiovascular disease in her father. The subject provided written informed consent for us to use un-identifying photos of her affected areas. Physical exam was notable for marked facial stubble subsequent to shaving  fingers. The changes were particularly impressive in the index and middle fingers on both hands Figure . An exte0 fingersSince androgens mediate all their cellular biological effects through its androgen receptor (AR), a member of the nuclear steroid receptor superfamily , we assuThe study was designed to evaluate the presence of androgen receptors in healthy female nail-bed cells. In this study we examined 5 healthy women between the ages of 20 to 50 years old. Inclusion criteria were: not currently pregnant or breastfeeding and greater than 6 months post-partum, post-abortion, post-pregnancy, or post-lactation at the time of collection. Participants must not have had any significant medical history or have taken any medications for three months prior to specimen collection. After informed consent was obtained, the participants were instructed to clip their own fingernails adjacent tissues at home and keep them in a 10% formalin solution that was given to them. Participants scheduled a time to drop off these tissues with the study coordinator. The nail bed tissues were processed for androgen receptor (AR) immunohistochemistry using standard protocols.Tissues were briefly dehydrated and embedded in paraffin. The blocks were sectioned at a thickness of 5 μm for the immunohistochemical staining; the primary AR antibody was used at 1: 5, dilution. After washing with DPBS, a biotinylated secondary antibody (diluted 1: 200), was used for 10 min, following streptavidin-conjugated peroxidase for 15 min. The sections were stained with diaminobenzidine (DAB) substrate and counterstained using hematoxylin. Immunohistochemical (IHC) stains are important and useful in the pathological diagnosis of Sertoli- Leydig cell tumors, as the various cell types may be difficult to recognize on H&E (Hemotoxin and Eosin) stains alone. The IHC stains not only help verify the type of the tumor cells that are present, but also help to differentiate them from other tumor types that may share similar histologic features. For example, they can differentiate between tumors with malignant behavior (i.e. carcinoma) from the benign behavior of the Sertoli-Leydig cell tumor. Sertoli cells typically are cytokeratin+, and EMA-, whereas Leydig cells are both cytokeratin- and EMA-. Both cell types show a Calretinin+, CD99+, Inhibin + phenotype.Post-op lab results revealed a swift and dramatic reduction in testosterone and estradiol to almost undetectable levels Table . During Following our novel observation of nail changes with the hyperandrogenemia in a post menopausal SLCT patient, we evaluated the presence of androgen receptors (AR) in the nail bed of normal healthy women. As shown in Figure Most Sertoli-leydig cell tumors are unilateral and occur between the ages of 25-30, with an average age of diagnosis at 25 years. Around 50% of these cases come to clinical attention due to the progressive masculinization as seen in our patient. In a post-menopausal female, mild hirsutism is generally idiopathic. However, in a patient with increased levels of androgens and virilization, a work-up should be done to rule out an androgen-producing tumor of the ovaries or adrenal glands. An ovarian origin of the virilization can be suspected in the presence of elevated androgens with normal DHEA or a negative dexamethasone suppression test.Sertoli and leydig cells are incapable of synthesizing estradiol  which suThe patient’s unique nail changes pre-surgery (Al-Hendy nail sign) and the subsequent reversal in nail curvature post-surgery reveals that androgens might have a direct effect on human nails and nail bed tissues. Other causes of spooning include iron deficiency anemia and hemochromatosis. However, we found the patient’s CBC and ferritin to be within normal reference range. The conclusion that androgens are very likely the causative agent behind these unique nail changes are based on the observation that these changes started to resolve after the normalization of serum androgen levels postoperatively in this patient. Similar to other steroids, androgens act on target cells by diffusing through the plasma membrane and binding to specific intracellular/nuclear receptors . Since aIdeally, we would have liked to study the expression of androgen receptor in the index case herself. Several limitations prevented us from accomplishing that. First, the patient was not easily accessible due to an extensive travel schedule. Additionally, due to the novelty of the observation, we did not know for sure if the nail changes were due to androgen excess until the changes began to resolve after the surgical removal of the SLCT tumor. Androgen, like other steroid hormones, induce its own receptor and hence measuring AR levels after surgery would not have reflected the status before surgery during the severe hyper-androgenemia phase. Future studies will address these limitations.Sertoli-Leydig cell tumors are a rare ovarian sex-cord tumor that usually occurs unilaterally. The diagnosis should always be considered in a young female that presents with symptoms of progressive virilization and ovarian mass on examination. However, albeit rare, this diagnosis should also be considered in post-menopausal females with similar symptoms. Detailed imaging studies should be promptly performed to search for the malignancy. Management of ovarian SLCT remains challenging due to lack of standard management protocol. Fortunately, since the vast majority of SLCTs are unilateral and confined to the ovaries in 90% of cases, surgical resection is generally curative. Prognosis is related to the tumor staging. The overall 5-year survival rate for stage I is 95% whereas stages III and IV is nearly 0%. Long-term follow-up is advised in all patients. This case revealed a unique and novel finding of nail changes associated with hyperandrogenism which demonstrates that androgen receptors can be found in the female nail bed. The clear connection of specific finger nail changes (Al-Hendy nail sign) and hyperandrogenemia awaits further confirmation from similar cases to reveal if this is limited to postmenopausal hyperandrogenemia or if it is also evident in reproductive age women. If confirmed to be present widely, this nail sign which is easy to detect, might function as a simple non-invasive screening tool for easy and early detection of hyperandrogenic endocrinopathies in women. Future studies looking at the effects of androgens on nails are warranted.All patient information used in the manuscript were de-identified and anonymized. A written consent was obtained from patient to use un-identifying photos of affected areas in this publications. Formal IRB consent was collected from subjects donating tissues for the androgen receptor studies.


We describe the clinical findings in two patients with pathologically diagnosed olfactory neuroblastoma (ONB) of the sinonasal area and the surgical methods used for its treatment. Using an endoscopic endonasal approach (EEA) without dura resection, along with radiotherapy, we successfully treated ONB at the Kadish stage A. One of our patients, however, experienced tumor recurrence 24 years after open surgery with radiotherapy that was conducted at another hospital. This patient was no longer eligible for radiotherapy, and the tumor was therefore resected with dura resection using an EEA combined with duraplasty. The dura resection with duraplasty using fascia lata and a pedicled nasal septal flap was minimally invasive. As with surgery without duraplasty, a postoperative computed tomography (CT) examination revealed that EEA with duraplasty led to quick improvement of the postoperative inflammatory response as well as pneumocranium. Here, we investigated whether to modify the method of surgery depending upon the primary site of early-stage ONB. We suggest that, in early-stage ONB, an endoscopic endonasal approach is an effective and less invasive method. It is also advisable to perform dura mater resection of the lesion site despite the absence of obvious intracranial invasions in image findings. Recently, much progress has been made in the field of otolaryngology with regard to the use of endoscopic endonasal surgery. In recent years, this type of surgery has been successfully performed on patients with skull base tumors. It has also been reported that an extended endoscopic endonasal approach (EEA) is effective in the treatment of malignant sinonasal tumors such as olfactory neuroblastoma (ONB), chondrosarcoma, chordoma, early-stage squamous cell carcinoma, and adenocarcinoma . Here, wCase  1. A 42-year-old man presented with a chief complaint of right-sided epistaxis and nasal stuffiness. He had no notable medical history. An anterior rhinoscopy revealed a dark-reddish tumour occupying the right nasal cavity  image showed a mass with a polyp-like appearance in the right nasal cavity, arising from the cribriform plate. No bone destruction was found. A contrast-enhanced coronal CT image detected a strongly enhanced mass. A magnetic resonance image (MRI) T1-weighted axial image revealed a low-intensity signal , whereasExtirpation of the lesion by EEA was performed under general anesthesia. The tumor was resected from the base, the right ethmoid sinus was opened, and the middle nasal concha and superior nasal concha were removed. Intraoperative rapid diagnostic tests were performed as needed. The mucosa around the base of the tumor was then abraded from the base of the nose and the cribriform plate was removed. Following this, the dura mater was exposed  and the Case  2. A 67-year-old man was diagnosed with an ONB (Kadish grade A-B) in 1989. He underwent lateral rhinotomy followed by irradiation and regularly completed follow-up visits at another hospital. At the end of August 2012, or 24 years after the initial tumor, the patient was referred to us for nosebleeds resulting from a mass in his right nasal cavity  which made radiotherapy impossible; therefore, surgery was performed with a dural biopsy and duraplasty using fascia lata plus a pedicled nasal septal flap. First, biopsies were performed at 4 sites of about 15 mm surrounding the surgical stump; the safety margin was confirmed with intraoperative rapid diagnosis. The tumor with the mucosa around the base of the tumor was abraded from the mucosa at the base of the nose. The right anterior ethmoidal artery was clipped and cut to control blood flow from the mucosa at the cribriform plate to the dura mater.The mucosa at the cribriform plate was ablated, and the dura at the tumor base was resected . After cONBs are thought to arise from the specialized sensory neuroepithelial olfactory cells that are normally found in the upper part of the nasal cavity, including the superior nasal concha, the upper part of septum, the roof of nose, and the cribriform plate of ethmoid . Even thONB shows varying biological activity, ranging from indolent growth to a highly aggressive neoplasm . This tuThe current recommended treatment strategy is surgery and, in selected Kadish stage A cases, this is combined with radiotherapy. Radiotherapy is conducted before or after surgery, at the primary site and cervical lymph nodes. Adjunctive chemotherapy may be added to this treatment depending on the degree of differentiation of the tumor for Kadish stage B. As for Kadish stages C and D, the suggested treatment strategy is preoperative chemotherapy and/or radiotherapy followed by surgery .P = 0.0018) [Recent findings have shown that sequential resection with negative margins is equivalent to open en bloc resection  11–16].–16.11–16 0.0018) . Althoug 0.0018) . Further 0.0018) .It has been reported that olfactory neuroblastoma most commonly recurs within the first 4 years but can recur very late, for example, after 19.4 years in one case. There is currently no universally accepted follow-up regime, but even late recurrence of the disease is eminently treatable. Therefore, a protocol for lifelong follow-up with both clinical examination and serial imaging including the neck and entire intracranial compartment has been proposed .In the 2 cases included in the present report, we performed first- and second-line EEA treatment for early-stage ONB. In the first case, dura biopsy plus duraplasty was not performed because residual mucosa in the cribriform plate area was intact in the intraoperative quick diagnostic test, and the current benign course was achieved, with gamma knife treatment for 50 months. In contrast, the second case showed tumor recurrence 24 years after craniofacial surgery plus radiotherapy. This patient was no longer eligible for radiotherapy, and the tumor was therefore resected including dura biopsy using an EEA combined with duraplasty to avoid microinvasion of the skull. To prevent a postoperative leak, we chose a dual repair procedure  and usedIn this minireview, we examined whether the dura mater adjacent to the base of a tumor should be resected in EEA surgery for early-stage ONB. During surgery, it is most important to first determine a clear resection range and then to confirm this range in an intraoperative margin study. We believe that, during operations to treat recurrent ONB, the dura mater adjacent to the base of minimally invasive tumors should be resected.We believe that, in early-stage ONB, EEA is an effective option. Additionally, it is advisable to perform dura mater biopsy of the lesion site despite the absence of obvious intracranial invasion in imaging findings. We propose a protocol for lifelong follow-up with both clinical examination and serial imaging.


Most bone tumors that occur in the clavicle are malignant. A few giant cell tumors (GCTs) of the clavicle have been reported; however, the most appropriate operative method for this tumor has never been discussed.A 54-year-old man noticed enlargement of the proximal aspect of the right clavicle. A plain X-ray revealed lytic change and ballooning of the proximal end of the right clavicle. The tumor was isointense on T1-weighted magnetic resonance images and showed a mixture of low- and high-intensity areas on T2-weighted images without extension to the surrounding soft tissues. Bone scintigraphy showed strong accumulation , and positron emission tomography revealed strong uptake of fluorine-18-2-fluoro-2-deoxy-d-glucose  in the proximal part of the right clavicle. Because we could not completely exclude malignancy, an open biopsy was performed. Pathologically, the tumor comprised mononuclear stromal cells and multinuclear giant cells, resulting in a diagnosis of a GCT of the bone. Although curettage may be considered for such lesions (Campanacci grade II), we chose resection to minimize the chance of recurrence. The tumor was resected en-bloc with the proximal half of the clavicle. No postoperative shoulder disproportion was observed, and full range of motion of the right shoulder was maintained. The patient was satisfied with the surgical outcome . He returned to his original job as a land and house investigator without any signs of recurrence for 1 year after surgery.Although GCT of the bone rarely occurs in the clavicle, the typical X-ray findings demonstrated in the present case are helpful for a correct diagnosis. Although en-bloc resection without reconstruction is appropriate for GCTs in expendable bones, there has been much discussion about shoulder function after total claviculectomy. Considering the importance of the function of the clavicle, which is to support the scapula through the acromioclavicular joint, we preserved the muscle attachments of the deltoid, trapezius, and pectoralis major. Because both the oncological and functional outcomes were satisfactory, we recommend preservation of as much of the clavicle as possible in patients with clavicular bone tumors. Giant cell tumors (GCTs) are aggressive bone tumors comprising osteoclast-like multinuclear cells and hyperplastic mononuclear interstitial cells. In the latest classification of bone tumors by the World Health Organization, GCTs are classified as “intermediate locally aggressive, rarely metastasizing” tumors . BecauseThis case has been reported in accordance with the Helsinki Declaration. This retrospective case report is an exemption by the ethics committee of Kagoshima University.A 54-year-old man noticed enlargement of the proximal aspect of the right clavicle. He made an appointment to undergo positron-emission tomography (PET)-based cancer screening 1 month later, which revealed a lesion with abnormal accumulation in the right clavicle. He was referred to our department for further examination. Plain X-ray revealed lytic change and ballooning of the proximal end of the right clavicle Fig. a, and thBased on the characteristic roentgenographic and CT imaging findings with ballooning of the affected bone, the primary differential diagnosis was a GCT. However, we could not completely exclude malignancy because of the affected site, patient age, and degree of accumulation on PET. We thus performed an open biopsy of the tumor. Preoperative angiography was performed to prevent dissemination due to massive perioperative bleeding; however, large nutrient vessels requiring embolization were not demonstrated. Fragile, yellowish-brown tumor tissue was obtained by the biopsy Fig. a. PatholBecause this was determined to be a grade II tumor  according to the Campanacci classification , curettaNo shoulder disproportion was observed postoperatively Fig. a. The raDahlin et al.  reportedA principal surgical treatment method for GCTs, which are benign bone tumors, is curettage. However, the relatively high local recurrence rate (>20 %) is a problem , 14, 15.Whether a clavicular bone tumor is malignant or benign is key to selecting the most appropriate surgical method. Obviously, total claviculectomy will be performed for malignant bone tumors , 20. HowIn conclusion, GCT should be considered when the typical X-ray appearance is observed in a patient with an aggressive clavicular bone tumor. If partial resection of the clavicle is necessary, we recommend preservation of as much of the clavicle as possible because no asymmetry, pain, or shoulder imbalance occurred in the present case.Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Series Editor of this journal.


Hamartomas are extremely rare splenic benign tumours in children. We present two cases, both in boys (6 and 8 years old), with left upper quadrant abdominal pain that were otherwise asymptomatic. Both patients showed a splenic mass on preoperative ultrasonography and magnetic resonance imaging (MRI). One patient had a focal splenic mass that was identified preoperatively with contrasted computed tomography (CT) scans. Both patients underwent a total splenectomy. Although multi-modality imaging findings were described preoperatively, the final diagnosis in each case was splenic hamartoma based on histology and immunohistochemistry. The postoperative courses were uneventful. Splenic tumours are relatively rare and include malignancies such as lymphomas, angiosarcomas, plasmacytomas, primary malignant fibrous histiocytomas, and metastatic disease. Benign splenic tumours are extremely rare and most are hemangiomas, cysts, and inflammatory pseudotumours ,2. Splenth postoperative day and reached 1,000 × 109/L on postoperative day 14. Postoperative anticoagulant prophylaxis with oral aspirin was initiated and continued for 4 weeks after surgery. Finally, the patient was discharged from the hospital on the 15th postoperative day. No complications developed during postoperative follow-up, and the patient was asymptomatic 5 years after surgery.An 8-year-old boy was incidentally diagnosed with a splenic mass by abdominal ultrasonography and admitted to our hospital for further investigation. He had no significant past medical history. The physical examination and the laboratory findings  on admission were unremarkable. Abdominal ultrasonography showed an isoechoic oval-shaped mass with smooth, well-defined borders in the mid-portion of the spleen that was 5.2 cm × 4.2 cm × 3.4 cm in size, within homogeneous echo Figure A. Colour9/L on the 7th day after surgery. Postoperative anticoagulant prophylaxis with oral aspirin was initiated and continued for 4 weeks after surgery. Finally, the patient was discharged from the hospital on the 13th postoperative day. No complications developed during postoperative follow-up, and the patient was asymptomatic 17 months after surgery.A 6-year-old boy was admitted to our department due to a 1-year history of left upper quadrant abdominal pain and worsening sickness for 1 week. Physical examination revealed a palpable spleen 3 cm below the costal margin. He had no significant past medical history, and the laboratory findings  were also unremarkable. Abdominal ultrasonography showed a hypoechoic mass with well-defined borders in the lower pole of the spleen that measured 9.4 cm × 8.9 cm × 6.4 cm in size, within heterogeneous echo Figure A. ColourSplenic hamartomas are rare benign tumours with a reported incidence of 3 in 200,000 splenectomies in a single centre series . The incAlthough most of the reports in the literature consist of adult patients ,8, smallAlthough the final diagnosis of splenic hamartomas is established by a pathological examination, a preoperative diagnosis using a combination of multi-modality imaging techniques may be possible ,12-14. OSplenic hamartomas should be differentiated from the more common neoplastic disorders of the spleen such as hemangiomas and ominous lesions of the spleen including primary haemangiosarcomas, lymphomas, and metastases . The maiIn conclusion, although splenic hamartomas are very rare tumours, they must be considered in the differential diagnosis of splenic lesions in children. However, a splenectomy may be necessary when malignancy cannot be ruled out preoperatively.Written informed consent was obtained from the patients for publication of this report and any accompanying images.The authors declare that they have no competing interests.LFZ and QQ drafted the manuscript. JFT, WX, WZG, and XHM also assisted with manuscript preparation. QQ revised the manuscript. All authors have read and approved the final manuscript.