Fibrous dysplasia is a benign tumour of the bones and is a disease of unknown aetiology. This report discusses a case of proptosis and visual deterioration with associated bony mass involving the right orbit.A 32-year-old Nigerian man of Yoruba ethnic origin presented to the eye clinic of our hospital with right-eye proptosis and visual deterioration of 7-year duration. Presentation was preceded by a history of trauma. Proptosis was preceded by trauma but was non-pulsatile with no thrill or bruit but was associated with bony orbital mass. The patient reported no weight loss. Examination of his right eye showed visual acuity of 6/60 with relative afferent pupillary defect. Fundal examination revealed optic atrophy. Computed tomography showed an expansile bony mass involving all the walls of the orbit. The bony orbital mass was diagnosed histologically as fibrous dysplasia. Treatment included orbital exploration and orbital shaping to create room for the globe and relieve pressure on the optic nerve.Fibrous dysplasia should be considered in the differential diagnosis of slowly developing proptosis with associated visual loss in young adults. Fibrous dysplasia is a benign, slowly growing disorder of bone in which the normal cancellous bone is replaced by immature woven bone and fibrous tissue . This coHistological examination provides the basis for an accurate diagnosis. The tumour is characterized by multiple small and irregular spicules of immature bone superimposed on a background of moderately cellular fibrous connective tissue . HoweverFibrous dysplasia may cause ophthalmic problems such as proptosis and dystopia, ocular motility problem and cosmetic deformity; however, visual loss represents the most common neurological complication of fibrous dysplasia affecting the skull .Fibrous dysplasia, though not rare, is a disease mainly documented among Caucasians ,3,8 and A 32-year-old Nigerian businessman of Yoruba ethnic origin was referred to the eye clinic of our hospital from another hospital in Nigeria with complaints of progressive protrusion of the right globe for 7 years. Six months prior to the onset of his complaints, he had hurt the edge of the right superior orbital margin against the edge of an iron bed at boarding school. No treatment was received for this. The protrusion of the globe continued to increase for about 7 months and then stopped. There was no pain and no diplopia but there had been progressive deterioration of the vision in the eye. Initial exploration of the right orbit performed at the referring hospital revealed a bony hard mass involving both the lateral and medial orbital wall. This mass could not be removed. There was no history of weight loss, heat intolerance or excessive weight gain. The proptosis was not made worse by the Valsalva manoeuvre. The patient experienced no unusual noises in the head. He had no history of swelling (of bony or soft tissue) in other parts of the body. Furthermore, there were no hoarseness of voice, dysphagia, cough, palpitation, headache, vomiting or seizure and no focal neurological deficits. The patient had no known hypersensitivities and no diabetes or asthma. He was single, the first of 6 children of his parents. His father had died at an age of about 62 years of an unknown cause, the mother was alive and well, aged about 60 years. There was no family history of similar eye problems.Examination revealed an otherwise healthy-looking man, with normal systemic examination. The right ocular examination revealed a visual acuity of 6/60 with a proptosis of 17 mm . The proptosis was non-axial , non-retropulsive, nonpulsatile, nontender and had no thrill and no bruit. There was chemosis of the overlying conjunctiva with moderate restriction of the extra-ocular muscle movement in all direction of the gaze. The pupillary reaction was sluggish with a relative efferent pupillary defect. Fundoscopy showed a pale disc with distinct margins. The left eye had a visual acuity of 6/5 with normal anterior and posterior segments. Cranial computed tomography scan Figures  showed aThe ophthalmic, neurosurgical and maxillofacial units of our hospital carried out a joint surgical exploration of the right orbit through a modified lateral orbitotomy, using an electric drill for the lateral orbital wall. Operative findings included thickened expanded zygomatic bone and greater wing of sphenoid, with the orbit being almost completely obliterated with expanded dense bony tissue. Gradual removal of the bony mass was performed in layers, using a hammer and chisel until a new orbital space was created. The histological finding Figure  was in kFibrous dysplasia results from a defect in osteoblastic differentiation affecting the final maturation of the bone . AlthougThe rapid worsening of visual acuity as described in this case could be as a result of cyst formation within the tumour, with resultant compression of the optic nerve and impairment of the venous return from the orbit. This is supported by the fact that there was lot of conjuctival chemosis which resolved after surgical decompression of the orbit. The loss of visual acuity may also have been a result of haemorrhage into the tumour resulting from the trauma sustained. However, this could not be substantiated from the histology results. Visual impairment following fibrous dysplasia has been attributed to many factors which include optic-nerve traction due to proptosis, sinus mucocele formation with raised intra-orbital pressure, haemorrhage within the tumour, optic canal stenosis, as well as cyst formation within the lesion .Establishing the diagnosis of fibrous dysplasia requires close cooperation between clinician, radiologist and pathologist which was demonstrated very well in the case reported. Orbital osteoma which is the most common benign tumour of the paranasal sinuses  may at tTherapeutic indication depends on the course of tumour and the development of complications. This could range from mere observation with serial radiological follow-up to medical therapy with systemic corticosteroid and surgical intervention. The surgical option adopted in this case met with the basic tenets of operative intervention using the treatment protocol proposed by Chen and Noordhoff . There wComplete resection of the lesion was not possible in this case because the entire posterior orbit was filled with the lesion. We concentrated on a curettage to provide enough room for repositioning of the globe, using a lateral orbitotomy approach which is associated with less morbidity and quick recovery. Cranio-orbital shaping is an acceptable mode of surgical treatment for fibrous dysplasia when it may not be possible to remove the pathological bone completely.Monostotoic fibrous dysplasia of the orbit causing neuro-ophthalmic complications associated with compressive mass effect should be considered in the differential diagnosis of slowly progressive proptosis in young adults.The authors declare that they have no competing interests.COB took part in the surgery and was a major contributor to preparing the manuscript. AOF, JKC and VNO took part in the surgery. OOK took part in the surgery and was a major contributor to preparing the manuscript. AOO performed the histological examination of the specimen. AIA and AMB were major contributors to the manuscript preparation. All authors read and approved the final manuscript.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.


Mesotheliomas are rare tumours that usually are seen in the pleura after asbestos exposure. Mesotheliomas have been reported around the testicular region but are even rarer following trauma, herniorrhaphy and long term hydrocoele.An elderly male farmer presented to us with an insidious onset of painless swelling in his left lower limb which gradually progressive. At the time of presentation it had involved his entire limb. A hard palpable mass of size 5 * 4 cms was detected in the left iliac fossa and a testicular enlargement was noted on the left side. The ultrasound of the testes showed that the left testis was enlarged 3.9*3*3.2 cms showing diffusely heterogenous echo-texture and irregular nodular surface with irregular hypoechoic thickening of the scrotal wall with left sided hydrocele. A separate hypoechoic *1.2 cms lesion was visualized in the anterior scrotal wall. FNAC from the scrotal mass showed tumour cells of simialr morphology present singly in monolayered sheets and in three dimensional fragments. The overall immunomorphological features suggested a malignant mesothelioma likely to have arisen from the tunica vaginalis.In conclusion, though a rare tumor, malignant mesothelioma of the tunica vaginalis of the testis should be considered whenever a paratesticular mass lesion is seen even without a history of trauma or asbestos exposure as is highlighted in this case. Ultrasound findings are helpful and fine needle aspiration of the tumor may assist in arrival at a diagnosis. Surgical orchidectomy remains the modality of treatment. Mesotheliomas are uncommon tumours that occur in the pleura or peritoneum usually following asbestos exposure which may be of any duration and sometimes several years in the past. They are not related to smoking and those originating in the tunica vaginalis of testis are rare, representing less than 5% of all mesothelioma . Trauma,A 65-year-old ambulatory male farmer from Bihar presented to our outpatient department with painless swelling of his left leg of two months that was insidious in onset and gradually progressive. The swelling started from his thigh and now involved the entire limb. He had no identifiable risk factors for a deep vein thrombosis. He had no bowel or bladder symptoms. The patient did not report any previous surgery or trauma to the region. He had worked as a hired farm worker all his life and gave no history of exposure to asbestos in his occupation.On examination he had stable vitals, and had pitting edema of the left leg. His right lower leg was normal. The ankle and knee joint were normal. No ulcers were seen on his leg. A difference in measurement between the two limbs of 5 inches over the thigh and 4 inches over the calf was noticed. He had inguinal lymphadenopathy on the left side, but no nodes were apparent to palpation on the right.A diagnosis of deep vein thrombosis and elephantiasis were entertained at initially. An examination of the abdomen revealed a hard palpable mass of size 5 * 4 cms in the left iliac fossa. This was a hard globular smooth nontender mass with lower margins disappearing below the inguinal ligament. On leg raising test the lump was found to decrease in prominence. The spermatic cord was palpated as a hard and cord like structure. The scrotal skin was normal. The left testis was found to be enlarged as a single hard mass of 10*5*4 cms. The testis was not tender and testicular sensation was preserved. The swelling was not fluctuant and not trans-illuminant. Per rectal examination did not reveal any abnormality.The initial diagnosis was now revised to suspect a testicular tumor – probably a seminoma or a lymphoma of the testes.The initial investigations revealed normal blood picture with a hemoglobin of 13 g/dL, a slight leukocytosis of 11200/cmm with neutrophilic predominance. He had normal renal and liver functions and the electrolytes were found normal. No abnormality was revealed on the chest radiograph and the color Doppler of the lower limb showed no evidence of deep vein thrombosis.The echocardiograph of the patient showed normal rhythm and no atrio-ventricular blocks were identified. The ultrasound examination of the abdomen and scrotum showed multiple well defined hypoechoic lymph nodes in the pre and para aortic regions along the left common iliac vessels. Right testis measured 3.2*2.1*2.4 cms and a right sided hydrocele was noticed. The left testis was enlarged 3.9*3*3.2 cms showing diffusely heterogenous echo-texture and irregular nodular surface with irregular hypoechoic thickening of the scrotal wall with left sided hydrocele. A separate hypoechoic lesion was visualized in the anterior scrotal wall. There was left sided hydrocele. The epididymis was normally visualized. The prostate was normal with a size of 2*3.5*2.7 cms.US guided FNAC from the left iliac mass were performed which revealed cells suggestive of a malignancy but were inconclusive for characterization.Computerized tomographic scan of the abdomen and the chest revealed few fibrotic lesions in Right Upper and apical segments of the right lower lobes of the lung. Multiple conglomerate lymphnodes (largest 5*4 cms) were seen in the retroperitoneal and pre pancreatic locations. A large (9*7*7 cm) lymph nodal mass was seen in the left hemi pelvis encasing left common iliac and left external and left internal iliac vessels. Pelvic and superficial inguinal lymphnodes (~2 cm) were also found to be enlarged. A simple cyst was visualized in the left kidneyAlpha fetoprotein was found to be 4.64 ng/ml  and beta human chorionic gonadotropin level was found to be 0.29 mIU/ml Trucut biopsy of the left iliac mass showed features of a squamous cell carcinoma. However, this was not consistent with the final diagnosis achieved after immunomorphological studies.Fine needle aspiration cytology from the scrotal mass was performed which showed tumour cells of similar morphology present singly in monolayered sheets and in three dimensional fragments. The tumour cells were polygonal in appearance with abundant cytoplasm and nuclei with vesicular chromatin, mild pleomorphism and eosinophilic nucleoli. The tumour cells were found positive for cytokeratin, epithelial membrane antigen (EMA) and calretinin. The overall immunomorphological features suggested a malignant mesothelioma likely to have arisen from the tunica vaginalis. The cytopathological findings of this case have been reported in detail by Mathur et al .Mesotheliomas of the paratesticular region usually present as tumors with no specific findings, have a broad age of distribution making pre-operative diagnosis difficult ,4. AlthoMesotheliomas are difficult to manage and no clear guidelines exist for management. Surgery has been suggested and orchidectomy is generally performed. Patients have been reported to have remained tumor free at 6 years following inguinal orchidectomy for limiIn conclusion, though a rare tumor, malignant mesothelioma of the tunica vaginalis of the testis should be considered whenever a paratesticular mass lesion is seen even without a history of trauma or asbestos exposure as is highlighted in this case. Testicular examination is usually neglected in medical departments while examining the abdomen and this oversight usually gives erroneous and misleading diagnosis. It is known that findings missed during the initial patient examination, remain mysteriously elusive during the further course of treatment. Ultrasound findings are helpful and fine needle aspiration of the tumor may assist in arrival at a diagnosis. Surgical orchidectomy remains the modality of treatment.Written informed consent for publication of this case report could not be obtained from the patient or from their next of kin. The patient underwent a high inguinal orchidectomy and then was lost to follow-up. Extensive attempts made to trace the patient proved futile. We believe this case report holds worthwhile clinical information which could not be communicated as effectively in any other way. We would not expect the patient or the family to object to publication.Since the time of submission it has come to the notice of the authors that the detailed cytopathological findings of the same case have been submitted by a separate set of authors in another publication.AG wrote the first draft of the manuscript. AA was involved in day to day patient care. RG provided insight and valuable inputs into the manuscript. SH delineated the radiological findings of the case. ABD was responsible for the overall care of the patient and the final decision on the patient management rested with him.


Spontaneous clostridium septicum infections are rare and are associated with a high mortality. Association of clostridium infection with colorectal malignancies have been previously reported and most cases are described in tumours of the ascending colon. We report our experience of clostridium septicum infection in the presence of tumour perforation in a series of two patients as a reminder of its association with sepsis in the presence of colorectal malignancy.We isolated clostridium septicum infection in a series of two patients admitted as emergencies. One patient was found to have a perforated caecal tumour intraoperatively whilst the other had a perforated rectal tumour. The clinical outcome and management of each case are reported and underlying reasons for variations in outcome are discussed.Although uncomman, the possibility of clostridium septicum sepsis should be borne in mind in patients who present with underlying malignancy and have sepsis. The cumulative effect of sepsis and malignant perforation is associated with a high morbidity and mortality. Awareness and early diagnosis of clostridium septicum may improve the prognosis of what is usually regarded as a fatal infection. Spontaneous Clostridium septicum infections are rare and are associated with cyclical neutropenia, diabetes mellitus and immunosupression -3. In adC.septicum sepsis is often fulminant with reported mortality rates of approximately 60% . Most ca9 (neutrophilia) and an elevated CRP. Urine microscopy revealed white cells and culture confirmed an E. coli urinary tract infection. She was treated initially with intravenous gentamicin, benzyl penicillin and metronidazole for an intra abdominal cause of sepsis according to the hospital microbiology protocol. The following day, she developed swinging pyrexia and blood cultures were taken. She proceeded to have a CT scan that revealed a distal sigmoid mass with extensive circumferential wall thickening and air beyond the bowel wall lumen suggesting localised perforation. There was no evidence of a pneumoperitoneum or of metastatic disease  adenocarcinoma. In view of the fact that she had a perforated rectal cancer, she was subsequently referred for adjuvant chemotherapy.A 80 -year -old woman was admitted as an emergency with a six-week history of progressively worsening right upper quadrant pain associated with nausea, vomiting and anorexia. She had not noticed any change in her bowel habit but had been using laxatives regularly.She had significant co-morbidities including Type II Diabetes mellitus, chronic renal failure for which she required erythropoetin injections, polymyalgia rheumatica, hypertension, ischaemic heart disease, deep vein thrombosis and gout. She had previously undergone a cholecystectomy and a total abdominal hysterectomy and bilateral oophorectomy.On examination, she had a temperature of 37.2°C and was tender in the RUQ. Initial investigations revealed a profound anaemia with haemoglobin of 5 g/l (hypochromic microcytic picture), an elevated white cell count and renal impairment. She had an isolated raised alkaline phosphatase and all of the other liver function tests were within normal parameters. An ultrasound scan of the abdomen revealed a possible ascending colon mass and she proceeded to have a CT scan to further evaluate these findings. CT confirmed an ascending colon mass with suspicious lesions in the liver suggestive of possible metastases.She underwent an emergency right hemicolectomy day 9 of admission. (Preoperatively a single dose of Gentamicin (7 mg/kg), benzylpenicillin 1.2 g and metronidazole 500 mg were administered. Intraoperatively, a perforated retroperitoneal tethered ascending colon tumour was found.In view of her significant underlying co-morbidities, she was transferred to the high dependency unit for postoperative care.Day 3 of her post-operative course was complicated by acidosis, worsening renal function, abdominal distention and worsening abdominal pain. She also developed a wound infection that became apparent the same day. An urgent CT scan without contrast was carried out which showed free fluid and pockets of air with in the fluid. No definite evidence of anastomotic leak was identified. In view of her deteriorating condition, an emergency laparotomy was performed. This was carried out 14 days later following her previous surgery. Areas of necrosis and pus were identified in the abdominal wall, which was debrided and tissue was sent for culture. Approximately 4 L of straw-coloured fluid was drained which was in keeping with ascites. There was no evidence of pus or anastomotic leak. A thorough washout was carried out and drains were placed. Tissue cultures of the abdominal wall showed a heavy growth of Coliform organisms and a moderate growth of positive for C.septicum and Faecal streptococcus. The tumour was noted to have perforated intraoperatively and these findings were also confirmed on histology and is likely to have been the origin of the abdominal wound infection. In view of this, vancomycin and metronidazole were used to treat sepsis. Her condition remained critical and she died of overwhelming sepsis 2 days later.Histology subsequently confirmed a Dukes C1 (pT4N1 Mx) mucinous adenocarcinoma of the ascending colon.Clostridium septicum is an anaerobic gram-positive spore forming bacillus and is a causative agent of gas gangrene or atraumatic myonecrosis . ProductAlthough C.septicum infection is an uncommon diagnosis, early identification, administration of appropriate antibiotics and surgery in which the sepsis was "resectable" improved outcome in case 1. In case 2 the patient had extensive abdominal wall necrosis and retroperitoneal sepsis that was not resectable and delay in diagnosis along with patient's significant co-morbidities contributed towards her poor prognosis.The possibility of C.septicum infection should be borne in mind in patients who present with an underlying malignancy and have signs of sepsis. Timely diagnosis is of the utmost importance for proper treatment. Cultures should be obtained to confirm the diagnosis and aid in future antibiotic regimens. Data from previous studies have shown that C.septicum is susceptible to a wide range of antibiotics such as clindamycin, penicillin and metronidazole . AlthougWritten 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 journalThe authors declare that they have no competing interests.MJC contributed to the study design, manuscript prerparation, editing and has approved the final version. JMM contributed to manuscript preparation. NNM was involved in the manuscript preparation and final editing. All authors approved the final manuscript.


Intracranial or spinal compressive lesions due to extramedullary hematopoiesis have been reported in the medical literature. Most of the reported cases are extradural lesions or, on rare occasions, foci within another neoplasm such as hemangioblastoma, meningioma or pilocytic astrocytoma. Often these cases occur in patients with an underlying hematological disorder such as acute myelogenic leukemia, myelofibrosis, or other myelodysplastic syndromes. Such lesions have also been reported in thalassemia major.We report the case of a 43-year-old Iranian woman in whom extramedullary hematopoiesis presented as a compressive cord lesion and then later as an intracranial lesion.To the best of our knowledge, we document the first reported case of sacral, lumbar, thoracic and cranial involvement in the same patient with extramedullary hematopoiesis, which seems both rare and remarkable. Intracranial or spinal involvement, manifesting as epidural lesions, due to extramedullary hematopoiesis (EMH) is rare. The intracranial lesions are also reported as foci within another intracranial neoplasm such as hemangioblastoma, meningioma or pilocytic astrocytoma ,2. ExtraOur case report, a 43-year-old Iranian woman, came to us complaining of back pain radiating to both of her lower extremities which had started two months previously. The pain was non-responsive to conventional medical treatments. In her general physical exam, mild splenomegaly was noted. A neuroexam showed decreased strength in both of her distal lower extremities (motor strength = 4/5) and absent deep tendon reflexes in her lower extremities. She had a limping gait due to pain. A sensory exam indicated problems in the L4 and L5 dermatomes. Her sphincter function was normal. A lumbosacral magnetic resonance imaging (MRI) scan showed an abnormal para-vertebral mass extending from L3 to S3 which became enhanced after a contrast injection Figure . In a do6/mm3; her haemoglobin level was 20.5g/dl; her hematocrit level was 67.3%; her mean corpuscular volume was 67 fl; her mean corpuscular hemoglobin level was 17.6 pg; her mean corpuscular hemoglobin concentration was 26.3g/dl; her reticulocyte level was 3.5%; her platelet count was 24,6000/mm3; her white blood cell count was 14.6 × 103/mm3; the percentage of polmorphonuclear cells in her blood was 46%; the percentage of monocytes was 2%; the percentage of band cells was 1%; the percentage of lymphocytes was 48%; and the percentage of myelocytes was 3%.Her red blood cell count was 7.66 × 10A consultation with the hematologist and a review of the peripheral blood smear yielded nucleated-RBC, anisocytosis (1+), microcytosis (2+), hypochromia (2+), poikilocytosis (2+), tear drop spherocytes and target cells Figure . Her Hb A two-week steroid therapy was of no benefit and her neurologic status deteriorated, so we planned to perform a surgical decompression. Since she manifested dominantly with lower motor neuron problems and the dorsal lesion was very extensive with a mild compressive effect over the cord, we decided to decompress the lumbar area. She was positioned prone and a bilateral laminectomy from L2 to S1 was performed. The extensive dark-grayish epidural lesion had a fragile consistency. We removed as much of the lesion as possible. The thecal sac and the roots were successfully decompressed. Microscopic studies showed considerable replacement of epidural fat with hematopoietic cells, predominantly from erythroid and granulocytic clones and also scattered megakaryocytes Figure .The adjuvant treatment continued with whole-spine radiotherapy, which was accompanied by her gradual recovery from paraparesis. Follow-up contrast studies undertaken every six months showed no evidence of any recurrence.Twenty-one months after her first visit, she came back complaining of headaches and visual blurring beginning two weeks previously. A clinical assessment revealed a grade 4 papilledema and mild paraparesis (muscle strength = 4/5) associated with generalized hyper-reflexia and extensor plantar reflexes.A whole axis contrast-enhanced magnetic study revealed a large extra-axial mass in her right frontal area, causing more than 10 mm midline shift, and a smaller mass in her left frontal area. The lesion became enhanced after a contrast injection and showed a dural tail sign. Patchy-enhancing foci was also evident in the meninges Figure . A spine6/mm3; her hemoglobin level was 11.9/dl; her hematocrit level was 37.6%; her mean corpuscular volume was 79 fl; her mean corpuscular hemoglobin level was 23 pg; her mean corpuscular hemoglobin concentration was 29g/dl; her platelet count was 78 × 103/mm3; her white blood cell count was 32.4 × 103/mm3; the percentage of polymorphonuclear cells in her blood was 71%; the percentage of lymphocytes was 13%; the percentage of monocytes was 5%; the percentage of myelocytes was 1%; the percentage of stab cells was 9%; and the percentage of metamyelocytes was 1%.Her red blood cell count was 4.76 × 10She underwent a platelet transfusion because of her thrombocytopenic state. According to consultation with the hematologist, her thrombocytopenia was explained by the hypersplenism.A course of steroid therapy was performed but it was unsuccessful. She showed signs of increased intracranial pressure, so we decided to proceed with surgery.To reduce the intracranial pressure, a right frontoparietal craniotomy and resection of the larger lesion was performed, which was accompanied with duraplasty to improve her clinical condition. During the operation, the mass appeared dark and vascular just below the dura mater and adhering to, and in some places invading, the pia matter. Immediately after the operation, she became hemiparetic on her left side and a CT scan showed severe edema compressing her right ventricle and no evidence of hemorrhage. The medical treatment for edema was initiated but in the second post-operative day, she became hemiplegic on her left side. The hemiplegia gradually and slowly improved, although modestly. A microscopic study of the lesion indicated erythroid, myeloid and megakaryocytic proliferation indicating EMH. After consultation with hematologists and oncologists, she was sent for cranial radiation. In her follow-up visit, six months after admission, there was no evidence of any radiologic recurrence of the supratentorial lesion and the patient's condition had improved. The follow-up visits continued every six months and after two years there had been no evidence of any radiologic or clinical recurrence and the patient's condition had improved.Involvement of the neuroaxis due to extramedullary hematopoiesis is not common. The reported cases occurred due to some major hematologic disorders such as myelodysplastic syndromes, acute myelogenic leukemia or thalassemia major ,2. In veSuggested treatments for these lesions include surgical removal and a combination of chemo-and radiotherapy in cases with hematologic malignancies, and blood transfusion when the underlying cause is thalassemia major, myelofibrosis or myelodysplastic syndromes.At the first presentation of our case report, her hemoglobin level was high. She had no prior history of smoking. Her erythropoietin level and arterial blood gas analysis were normal. A bone marrow biopsy did not indicate myelofibrosis or a solid cancer metastasis and she has been disease free for two years, so these diagnoses were ruled out. We did not find the cause of her high hemoglobin level; however, it was not seen in the follow-up laboratory tests.et al. although EMH has occurred in a malignant meningioma in his report [In our experience, she showed no sign of anemia to justify transfusion and surgical resection was considered necessary on both occasions due to the significant compressive effect and her deteriorating condition. Radiation as a treatment modality caused the thoracic lesion to disappear and there has been no evidence of any recurrence during follow-up checks. An increase of cerebral edema after surgical resection of these lesions has been reported by Gregorios s report .Although CNS involvement in EMH is very rare, this entity deserves attention as a differential diagnosis in patients with an underlying hematologic disorder.CT scan: computed tomography scan; EMH: extramedullary hematopoiesis; HCT: hematocrit; Hgb: hemoglobin; MCH: mean corpuscular hemoglobin; MCHC: mean corpuscular hemoglobin concentration; MRI: magnetic resonance imaging; Plt: platelet; RBC: red blood cell; U: unit; WBC: white blood cell.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.ASS analyzed and interpreted the patient data regarding the lateral sacral meningocele and performed the surgery. AS performed the review of literature and was a major contributor in writing the manuscript. All authors read and approved the final manuscript.


Mesothelial cell inclusions in lymph nodes are of rare occurrence and can be mistaken as metastatic adenocarcinomas, mesothelioma or sinus histiocytosis. These are usually found in mediastinal and abdominal lymph nodes and are associated with effusions. We report a case of benign mesothelial cell inclusions in cervical lymph nodes, which was associated with chylous effusion, and immunohistochemistry revealed unusual weak cytoplasmic epithelial membrane antigen positivity in the cells. Benign inclusions in lymph nodes are broadly classified as epithelial, nevomelanocytic and decidual. Mesothel19/L; platelets 2×109/L. Peripheral smear was normal. Serum lactate dehydrogenase (LDH) level was 360 IU/L (<380 IU/L). Alpha-fetoprotein (AFP), beta-human chorionic gonadotropin (β-HCG) and carcinoembryonic antigen (CEA) levels screened in the serum were within normal limits.A-16-year-old boy had presented with severe breathlessness, which was distressing even at rest. He had low grade fever and had a remitting and relapsing course. There was facial puffiness, and significant pedal edema. Chest radiograph revealed bilateral pleural effusion. Ultrasound examination of the abdomen revealed gross ascites; however, there was no organomegaly or any lymphadenopathy. Computerized tomography of the chest revealed pleuropericardial effusion with enlarged mediastinal, left supraclavicular and cervical lymph nodes, suggestive of lymphoma. His complete blood counts were: hemoglobin 15.2 g/dL; total leukocyte count 5.5×10Bone marrow evaluation was within normal limits. Both the ascitic and pleural fluids tapped were grossly milky white and sent for biochemical and cytological evaluation. Biochemical evaluation established the chylous nature of the fluid due to the presence of chylomicrons, markedly elevated triglycerides and normal cholesterol levels. Cytological evaluation revealed pleocytosis with the presence of benign lymphocytes and mesothelial cells only and was negative for malignancy.Left cervical level V lymph node was biopsied for conclusive opinion. Grossly, there were four nodes ranging from 1 cm in diameter to 2×1.5×1 cm. Cut section of all nodes revealed yellowish tan to gray white, smooth to mottled surface. Microscopically, there were small clusters and singly scattered, round to polygonal cells, seen in the subcapsular and interfollicular sinuses of the nodes . These cThe breathlessness of the patient was progressive and the patient collapsed due to cardiorespiratory arrest.Benign inclusions in lymph nodes are of various types. These are uncommon in occurrence and, thus, mistaken as metastatic carcinoma or melanomas depending on the type of inclusions.6 The difMesothelial cell inclusions are of very rare occurrence and usually involve the mediastinal and retroperitoneal groups of lymph nodes.–46 The i414The main problem with mesothelial inclusions is that these mimic sinus histiocytosis, metastatic adenocarcinoma, and metastatic mesothelioma.–35 The m3188The mechanism of the mesothelial cell inclusions is also distinct as compared to other inclusions. Developm1151Although the cause of death could not be established in our patient, we presume that some other undiagnosed pathology resulted in these mesothelial inclusions, which were an incidental finding. It is important to note that the presence of these benign and innocuous epithelial elements in the sinuses may lead to a misdiagnosis of metastatic carcinoma to the unwary.Mesothelial cell inclusions in lymph nodes are a rare occurrence and may be mistaken for malignant adenocarcinoma or mesothelioma. The cellular characteristics with the immunoprofile should be evaluated carefully to prevent such errors, even though these occur in rare sites.


Carcinosarcoma of the lung is a malignant tumor composed of a mixture of carcinoma and sarcoma elements. The carcinomatous component is most commonly squamous followed by adenocarcinoma. The sarcomatous component commonly comprises the bulk of the tumor and shows poorly differentiated spindle cell features. Foci of differentiated sarcomatous elements such as chondrosarcoma and osteosarcoma may be seen. Aspergillus pneumonia is the most common form of invasive aspergillosis and occurs mainly in patients with malignancy, immunocompromizing or debilitating diseases. Patients with Aspergillus pneumonia present with fever, cough, chest pain and occasionally hemoptysis. Tissue examination is the most reliable method for diagnosis, and mortality rate is high.We describe a case of primary carcinosarcoma of the lung concurrently occurring with invasive pulmonary aspergillosis in a 66-year old patient. Primary carcinosarcoma of the lung is exceedingly rare -8. In thInvasive pulmonary aspergillosis is a spectrum of reactions that depend on a combination of patient immunologic status, underlying lung condition and the nature of exposure to aspergillus fungus. It most often presents as aspergillus pneumonia and almost always involves immunoecompromized or debilitated patients with underlying malignancy . Acute lA 66 years old African American man presented to the hospital with 1 week history of progressive shortness of breath and bilateral calf pain. He complained of occasional productive cough but denied any chest pain, hemoptysis, night sweats, palpitation, or dyspnea. He had a history of peripheral vascular disease and prostate cancer Gleason's score 6(3+3) about 8 years ago for which he had prostatectomy and subsequent penile implant for erectile dysfunction. He had an extensive smoking history but no alcohol or street drug abuse. Furthermore, he had a prior 8-year history of incarceration and a family history of lung cancer.Due to his chest symptoms, he had a chest x-ray followed by Computerized Tomography (CT) scan of the chest which showed a left upper lung mass (4.5 × 5.5 × 5 cm) with mediastinal and right hilar adenopathy Fig.. No pleu3, and absolute neutrophil, monocyte and lymphocyte count of 4.6 k/mm3, 0.6 k/mm3 and 3.2 k/mm3 respectively. Serum creatinine was 1.4 mg/dl and blood urea nitrogen 14 mg/dl. HIV and Hepatitis C serology were negative. He improved and was discharged on voriconazole. However, he presented again after about 8 weeks with new onset hemoptysis and night sweats. He subsequently had bronchoscopy with bronchoalveolar lavage (BAL) which returned negative for mycobacterium, fungus, legionella and cytomegalovirus on culture. Direct Fluorescent Antibody of BAL fluid was negative for Parainfluenza 1, Adenovirus, Herpes Simplex I&II, Respiratory Syncytial Virus, Varicella Zoster Influenza A&B and Adenovirus. BAL fluid was negative for malignant cells and Pneumocystis carinii.A CT guided fine needle aspiration cytology of the left lung mass showed inflammatory necrotic background with several large aggregates of fungi. On Gomori Methanamine Silver (GMS) stain, the hyphae had uniform diameter, septation and branching at 45 degree, morphologically compatible with aspergillus species Fig. . A speciPulmonary function test showed an obstructive pattern (FEV1/FVC ratio 58% of reference). He subsequently had a thoracotomy with a left upper lobectomy revealing biphasic malignant tumor (carcinosarcoma).A left upper lobectomy (20 × 15.5 × 5.5 cm) was done. Sectioning revealed a large tan-white circumscribed partly hemorrhagic mass with central necrotic cavity. The mass was abutting the pleural surface and measured 8.5 × 6.5 × 5.5 cm of which intra-operative frozen section was diagnosed as poorly differentiated squamous cell carcinoma. Interestingly, final surgical pathology examination revealed a poorly differentiated biphasic malignant neoplasm with epithelial and spindle cell components and necrosis Fig. . The carWithin the resection specimen areas of infarction and coagulative necrosis were identified, some of them bordering viable tumor Fig. . In one Additionally, intraoperatively obtained pleural fluid showed many malignant cells arranged in sheets and single cell pattern. The tumor cells were round to spindle and highly pleomorphic. Immunostains for CK 5/6, CK 7, CK 20, TTF-1, CD 15 and CD 30 were all negative in the tumor cells.Unfortunately, our patient had a complicated postoperative course in the intensive care unit. He developed pneumonia with Acinetobacter and was difficult to wean him from the ventilator. He finally succumbed to cardiac arrest.Pulmonary carcinosarcoma is a malignant tumor with a mixture of carcinoma and sarcoma ,2. In thThe carcinomatous component is more often squamous cell carcinoma, followed by adenocarcinoma and large cell carcinoma; whereas the most common mesenchymal component is poorly differentiated spindle cell sarcoma. Nevertheless, foci of rhabdomyosarcoma, osteosarcoma, and chondrosarcoma are often found .In a retrospective analysis of 2,400 lung cancer patients between 1975 and 1995 conducted by Huwer H et al, only seven patients (0.3%) had pulmonary carcinosarcoma [Immunohistochemical (IHC) staining enhances the distinction of carcinomatous from sarcomatous components within the tumor. When heterologous sarcoma elements such as cartilage or skeletal muscle are present, it is easier to confirm the biphasic nature of the tumor, although immunostains can be of further help such as Myogenin and Myo D1 (rhabdomyosarcoma); smooth muscle actin and desmin (leiomyosarcoma); and S100 (chondrosarcoma). However, carcinosarcomas are difficult to diagnose preoperatively. Biopsy of the tumor, especially when centrally located, often shows only one component, and peripheral tumors are difficult to reach endoscopically .Histogenetically, Carcinosarcomas may represent malignant epithelial neoplasms undergoing divergent tissue differentiation originating from a single clone . The proInvasive Aspergillosis, is a major cause of morbidity and mortality in the severely immunocompromised. Risk factors include prolonged and severe neutropenia, hematopoietic stem cell and solid organ transplantation, advanced AIDS, and chronic granulomatous disease . It is rNivoix Y et al retrospectively analyzed 289 patients with presentation that fulfilled the criteria for possible, probable, or proven invasive aspergillosis according to the international definitions. The predictors of increased overall mortality were in patients who received allogenic hematopoietic stem cell or solid-organ transplant, prior respiratory disease, corticosteroid therapy, renal impairment, low monocyte counts, disseminated aspergillosis, diffuse pulmonary lesions, pleural effusion, and proven or probable aspergillosis .In our case, the invasive aspergillosis and the subsequently diagnosed carcinosarcoma occupied the same location of the left upper lobe. The typical tissue reaction in aspergillus pneumonia is known to be hemorrhagic infarction with a sparse inflammatory infiltrate , and ourIn conclusion, the combination of invasive aspergillosis and solid malignancies is an extremely rare occurrence. To the best of our knowledge, there has not been any reported case of pulmonary carcinosarcoma and invasive aspergillosis in the literature. Furthermore, this case report is significant because the patient did not have any of the commonly described risk factors for developing invasive aspergillosis. Limited studies found in literature have shown poor prognosis in patients with either invasive aspergillosis or pulmonary carcinosarcoma. Whether the coexistence of these two entities in a patient increases mortality remains to be determined in case series, retrospective or prospective studies.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.The authors declare that they have no competing interests.AO provided the clinical data and drafted the manuscript, DD described the pathology component and took photographs, participated in writing the discussion, coordinated and edited the manuscript, WH edited the clinical part of the manuscript, TV provided part of the references and edited the clinical case presentation, HS reviewed the entire manuscript, participated in writing the discussion and the pathology component and edited the manuscript. All authors read and approved the final manuscript.


Primary liver tumors in children are rare with malignant hepatoblastoma being the most common neoplasm. In this report, we describe the diagnosis and clinical management of a large liver tumor in a 3-year-old child that displayed the features of both, conventional hepatoblastoma and malignant teratoma. Pathological assessment on a pre-operative bioptical specimen showed an immature teratoid tumor with no area of hepatoblastic differentiation present. Histological and immunhistological examination of the resected tumor specimen additionally showed tumor areas of very different differentiation pattern intermixed with each other, namely areas of hepatoblastoma-typical and neuroblastoma-like morphology as well as areas of rhadomyosarcomatous differentiation.After chemotherapy the tumor size increased and an extended right hemihepatectomy was performed. Post-operatively, the general condition of the child improved and adjuvant chemotherapy was started two weeks later. 36 months after initial diagnosis the patient is healthy, in good general condition, and without any sign of residual tumor disease.Overall, we describe the diagnosis and clinical management of a large liver tumor in a 3-year-old child that displayed the features of both, conventional hepatoblastoma and malignant teratoma and was designated as mixed hepatoblastoma and teratoma. Though mesenchymal tumor portions can occur within hepatoblastomas, most commonly osteoid or chondroid, our case is different as it presents a large spectrum of mesenchymal and epithelial differentiation pattern in most of the lesion. Primary liver tumors in children are rare. The liver is embryologically derived from both meso- and endodermal tissues and, thus, can develop a wide variety of both benign and malignant neoplasms. Hepatoblastoma is the most common malignant hepatic neoplasm of childhood, representing about 75% of the primary liver tumors in children in Western countries. Hepatic  teratoma is an extremely rare tumor with only single cases reported world-wide -4. In thA critically ill 3-year-old boy presented with a hard mass beneath the right costal margin. Magnetic resonance tomography showed a large, inhomogeneous mass measuring 10 × 9.5 × 7.3 cm with an intra- and extra-hepatic portion fig.  and a paPathological assessment on a bioptical specimen showed an immature teratoid tumor with no area of hepatoblastic differentiation present. After a 5-day PEI chemotherapy following the MAKEI 96 protocol of the German Society for Pediatric Oncology and Hematology (GPOH) [The patient recovered well and showed no signs of further tumor disease 36 months postoperatively.Histological examination showed tumor areas of very different differentiation pattern intermixed with each other (i. e. not representing a collision tumor). Roughly, a fifth of the examined tumor displayed a hepatoblastoma-typical morphology partly resembling embryonic and fetal hepatoblastoma fig. . About 1Immunohistochemical analysis  revealed expression of HepPar-1 selectively in the hepatoblastic cells fig.  includinFISH analysis demonstrated no amplification of the MYCN-oncogene, which would have been an indicator of a poor prognosis.Post-operatively, the general condition of the child improved and the first of four 10-day IPA chemotherapy cycles following the HB 99 protocol of the GPOH was started two weeks later. 36 months after initial diagnosis the patient is healthy, in good general condition, and without any sign of residual tumor disease.Hepatoblastoma is a rare, but the most common hepatic malignancy of childhood with a peak incidence from 6 months to 3 years. The etiology of hepatoblastoma is unknown, but it has been associated with Wiedemann-Beckwith syndrome, familial adenomatosis polyposis coli, prematurity and low birth weight. Hepatoblastomas are well-defined, solid, mostly intrahepatical lesions. Lymphatic and hematogenic metastases occur in advanced disease stages with the latter mostly involving the lungs. AFP levels are nearly always elevated in hepatoblastomas and are correlated in most cases with the stage of the disease [Teratomas are rare neoplasms (incidence 0.7/100.000 children/year) with tissue derivatives of all three germ layers. Teratomas mostly occur in the ovaries, the sacrococcygeal region, the testes, and the central nerval system and only rarely in other locations with less than 5% occurring in the abdomen .Our case represents a nearly unique combination of both tumor entities, hepatoblastoma and malignant teratoma in a young boy. Though mesenchymal tumor portions can occur within hepatoblastomas, most commonly osteoid or chondroid , our casThe treatment of the presented mixed hepatoblastoma and teratoma of the liver was based on a combined systemic chemotherapy and surgery. Therapies of such a mixed tumor tissue have not been described yet, but children with hepatoblastomas have nowadays, due to the new therapy modalities, a rather good prognosis with a 5-year survival rate of over 70%  with curIn summary, we report on the diagnosis and clinical management of to our knowledge the second case of a mixed hepatoblastoma and teratoma in a young boy . ClearlyThe authors declare that they have no competing interests.AM collected data and was the main author of the manuscript. AK helped with evaluation of the data. UB was the treating pediatric oncologist, HT the operating surgeon, WH the responsible radiologist, IL the consiliary pathologist, NS helped writing the paper and CW and TA were the local pathologists making the primary diagnosis. TA was the senior supervisor of the work performed. All authors critically reviewed the manuscript.


The most mobile organ of the body, the tongue is associated with various congenital anomalies; most of which are in association with many other systemic abnormalities. Rarely do they occur in isolation. Isolated aglossia, that we presented, is one of the more rare presentations.Our patient is a 6-year-old male child of Asiatic origin from Kashmir (India), who was physically well built and mentally sound and presented with history of impaired speech. The patient had normal velopharangeal competence but absence of tongue which was replaced by a small mucus membrane projection near the floor of oropharangeal isthmus. The patient had no difficulty in feeding or taste sensation but he was unable to pronounce lingual consonants.Isolated aglossia is very rare condition explained on the basis of growth failure of lateral lingual swellings and tubercular impar. Such patients do not usually need reconstruction of tongue; as feeding, swallowing and taste sensations are usually intact and speech cannot be improved by reconstruction. However, malocclusion of teeth needs to be taken care of. The tongue is the most mobile organ of the body, which helps in swallowing and speech. It has a special property of taste sensation and in addition helps in normal development of teeth and mandible. Various inherited, congenital, and developmental anomalies of tongue have been reported. Some are of minor clinical significance like ankyloglossia, fissured or scrotal tongue, and lingual thyroids. Some are of major severity and cause discomfort to person and affect their normal living pattern. Hypoglossia or microglossia is one of the more rare congenital anomalies manifested by the presence of a small or rudimentary tongue. Aglossia, the complete absence of tongue since birth, has also been reported in literature . Most ofA 6-year-old male child of Asiatic origin from Kashmir (India) presented to our department with history of impaired speech. On detailed examination he was found to be physically well built and mentally sound. Local examination revealed normal velopharangeal competence but with absence of tongue which was replaced by a small mucus membrane projection near the floor of oropharangeal isthmus. Patient had no difficulty in feeding or taste sensation but he was unable to pronounce lingual consonants. He had hypoplastic mandible with class 2 malocclusion . There wRadiology revealed hypoplasia of the mandible with malalignment of teeth .Aglossia is a known entity but is usually associated with various craniofacial and limb abnormalities. Hall has given the following detailed classification for such anomalies .As per this classification, our patient is catagorized as Type-1B. Various congenital malformations along with aglossia have been reported. Preis et al noticed anodontia, epidermoid of right eye, 6th and 7th nerve palsy, left hypodactyli and VSD along with aglossia . KantapuIsolated aglossia is a rarity, reported only by few ,9. Our pIt is concluded that isolated aglossia is very rare condition explained on the basis of growth failure of lateral lingual swellings and tubercular impar, the actual etiology of which remains largely unknown . Such pa


To evaluate the role of diffusion-weighted magnetic resonance imaging (MRI) and proton magnetic resonance spectroscopy (MRS) in the diagnosis of different orbital masses and their advantages over conventional MRI.The study included 20 patients presenting with proptosis. Every patient was subjected to thorough clinical examination, conventional MRI “T1 weighted, T2 weighted, and postcontrast T1 weighted if needed,” diffusion-weighted MRI, and proton MRS. Orbitotomy was performed, the orbital mass was excised, and histopathological examination was performed.Diffusion-weighted MRI could differentiate between benign lesions and malignant tumors in 70% of cases; however, overlap occurred in 30% of cases with benign tumors showing restricted diffusion whereas proton MRS could differentiate between benign and malignant tumors in 90% of cases.in vivo tissue characterization. Magnetic resonance spectroscopy seems to be the more accurate modality.Diffusion-weighted MRI and proton MRS can potentially increase the accuracy of diagnosis of orbital masses through  N-acetyl aspartate, choline, creatinine, and lactate in the tissue examined.Proptosis is one of the most common indications for an ophthalmologist to order neuroimaging. The two imaging techniques for the brain and orbit are computed tomography and magnetic resonance imaging (MRI) scans.Twenty patients with proptosis were included in this clinical study. They were randomly selected from patients with proptosis attending the outpatient clinic of Mansoura Ophthalmology Center. This study was approved by the Human Subjects Committee of the University of Mansoura, and adhered to the Declaration of Helsinki. Written informed consent was obtained from all participants.Patients who had bony lesions were excluded from the study. Diagnosis of proptosis was made when patients had readings more than 20 mm using Hertel's exophthalmometer. All patients underwent thorough ophthalmological examination including history taking, assessment of visual acuity and degree and direction of proptosis, measurement of intraocular pressure (IOP), and fundus examination.Conventional MRI, diffusion-weighted MR imaging (DWI), and MRS of both orbits were done to all patients. Examination was done in MR Unit, Mansoura University Hospitals, using 1.5 T Siemens system equipped with echoplanar capabilities (Symphony).b values of 0, 500, and 1000 s/mm2.Diffusion-weighted MR imaging was obtained using a multisection single-shot echo planar imaging sequence (TR/TE/NEX: 2200/139 ms/1) with X, Y, and Z). Sections of 5 mm thickness, interslice gap of 1 mm, FOV of 220-240 mm, and 128 × 256 pixel matrix were used for all images. Scanning time was less than 2 min.The diffusion gradients were applied sequentially in the three orthogonal directions (b = 1000) was classified as hypointense (free diffusion) or hyperintense (restricted diffusion).Three types of images were obtained: orthogonal images, trace images, and apparent diffusion coefficient (ADC) maps. The signal intensity of the lesion on DWIs  of the lesions and the mean ADC value was calculated. The ADC values were expressed in 10Proton MRS was performed with either chemical shift imaging (CSI) or single voxel spectroscopy (SVS) depending primarily on the appearance of the lesion in the preceding MR images. Four patients underwent SVS examination as they had homogenous, well-defined lesions with no infiltration of surrounding tissues, while the remaining 16 patients underwent CSI.Water suppression of the dominant water signal by the CHESS technique, outer volume fat suppression as well as magnetic shimming were performed automatically for all patients at the beginning of both SVS and CSI examinations. Curve fitting was performed automatically for all obtained spectra. The criteria for determining the presence of choline in a lesion were the appearance of a clearly identifiable peak at 3.2 ppm spectra. Peak height provided the level of concentration of choline. Any other peaks were not considered as they were not significant in the differentiation between benign and malignant lesions.Final diagnosis of all orbital masses was made by histopathological examination after orbitotomy. Correlation among the DWI, MRS, and histopathological results of all orbital masses was performed.Twenty patients with orbital soft tissue masses were included in this study. They were 10 men and 10 women. Histopathologically, 14 cases had benign lesions whereas the remaining six had malignant lesions. The classification of lesions in the study based on histopathology is presented in On DWIs, 57% of benign lesions appeared hypointense indicating free diffusion. A case of cavernous hemangioma, the two cases of optic nerve glioma, two cases out of five of idiopathic orbital inflammatory disease, and one case out of three of dysthyroid ophthalmopathy appeared hyperintense indicating restricted diffusion. The ADC was high for all benign lesions except for these six cases which had low values. All (100%) malignant tumors appeared hyperintense indicating restricted diffusion and showed low ADC values .−3−3mm2/s. Histopathology of this case is shown in Diffusion-weighted MRI could differentiate between benign lesions and malignant tumors in 70% of cases. However, overlap occurred in 30% of cases with benign tumors showing restricted diffusion. b = 1000) the lesion is hyperintense (restricted diffusion) and ADC map shows a low ADC value of 0.72 × 10−3 mm2/s In a case of cavernous hemangioma in the right orbit of a 54-year-old man, the axial and coronal fast spin echo show a well-defined homogeneous intraconal mass surrounding the optic nerve which appeared displaced medially and inferiorly with subsequent proptosis. Despite its benign nature, in axial DWI , and was present in all malignant tumors (100%) . Chemicain vivo with DWI. This measurement of the self-diffusion coefficient of water indicates the mobility of water within tissue and is called the apparent diffusion. In DWI, a pair of pulsed magnetic field gradients is applied and water molecules that have diffused during the time interval between the applied pulses show a larger signal loss than water with restricted motion. Restricted diffusion appears bright on DWI. Such imaging can be performed without the need for the administration of exogenous contrast medium. It yields quantitative and qualitative information that reflects changes at the cellular level and indicates the integrity of cell membranes.et al.et al. reported that the brightness of the lesions were higher and the ADC values were lower in the following order: orbital lymphoid lesions; orbital inflammatory syndrome; and orbital cellulites.15Although abnormalities of diffusion are generally not pathognomonic, diffusion MRI affords information about tissue changes for specific disorders that complement information obtained with standard MR techniques and frequently shows pathology earlier.N-acetyl aspartate (NAA), and (4) lactate. Reduction of NAA on MRS is a marker of neural loss. Certain tumors have no NAA  or markedly decrease NAA . Creatine can be increased in hypometabolic state caused by ischemia or tumor , or it may be decreased in hypermetabolic states. Choline is a component of cell membranes, and increased choline might suggest increased membrane synthesis in an active proliferating or a solid, hypercellular tumor. Creatine often remains stable in other disease processes and can be used as a control for MRS with levels of other metabolites expressed as a ratio to Cr.16Magnetic resonance spectroscopy is based on detecting various proton MR spectra. The four major resonances for MRS are: 1) choline-containing phospholipids, (2) creatine and phosphocreatine, (3)  choline-AA), and  lactate.in vivo physiological and metabolic information, complementing morphologic findings from conventional MRI in the clinical setting. Both techniques are noninvasive techniques to monitor changes in the biological structure of tumor tissue. They are a potential diagnostic tool for in vivo tissue characterization. The combination of MRS and MRI has led to mapping metabolites from normal and neoplastic tissue within the time limit of a routine study. The applications of these newer techniques are a clinical aid to the ophthalmologist for in vivo diagnosis of certain diseases.Advanced MRI techniques, such as MRS and DWI, can provide


Renal AML complicating tuberous sclerosis shows a rapid growth and its rupture is frequently associated with hemorrhagic shock as a result of profuse retroperitoneal bleeding, necessitating an aggressive therapeutic approach. This report describes the long-term clinical progress of 28 year-old woman with tuberous sclerosis with a ruptured giant AML that underwent unilateral nephrectomy, who has been followed up after treatment with concomitant application of computed tomography during selective arteriography to conserve the remaining normal renal parenchyma, and in whom the need for initiation of dialysis has been successfully avoided, with conspicuous reduction of the tumor size. AML of the kidney is a benign neoplastic lesion that is composed of vascular and smooth muscle components and adipose tissue, and has come to be recognized as a rather common complication of tuberous sclerosis ,2. AML cThe patient, a 28-year-old Japanese woman, had been under periodic follow-up since her childhood for tuberous sclerosis, and underwent right nephrectomy for AML at the age of 17; she continued to be under follow-up for immediate detection of AML developing in the remaining left kidney. During the course of follow-up, the patient developed sudden abdominal pain and needed emergency hospitalization because of decreased serum Hb level. Laboratory examination at admission revealed a serum Hb level of 7.3 g/dL and serum Cre of 0.9 mg/dL.Abdominal CT revealed a fairly large-sized tumorous lesion situated primarily in the left retroperitoneum and containing a huge aneurysm-like hemorrhage. The left renal artery was dilated to nearly the size of the aorta . While aAn abdominal angiography revealed marked distension of the left renal artery, and it was difficult to visualize the mass clearly and to accurately delineate the vascular supplies to the normal kidney parenchyma, even by left renal arteriography  . Based oAt half a year and about 5 years after the embolization, CT scans showed marked contraction in the size of the tumor and hematoma, with no evidence of any fresh hemorrhage , and 5d.Tuberous sclerosis is frequently complicated by AML, which is known to occur more frequently as multiple lesions and to grow to larger sizes as compared with idiopathic AML. It has also been documented that AML associated with tuberous sclerosis shows a high likelihood of rupture during the clinical course, with resultant hemorrhagic shock secondary to hematuria and retroperitoneal bleeding -10. In rIn the present case, the attempt to delineate the whole mass by angiography was virtually unsuccessful even after injection of 35 mL of contrast medium at the rate of 7 mL/sec. Therefore, we employed CTSA, which enabled efficient differentiation between the feeding arteries to the tumor and the normal renal vasculature with the use of a smaller volume of contrast medium early after the start of treatment, and thereby, efficient embolization therapy. Marked amelioration of the abdominal compressive symptoms was also anticipated with the expected contraction of the retroperitoneal hematoma with the ruptured AML and reduction in the diameter of the tumor. We used metallic coils in this case, but the use of metallic coils calls for discretion, that is, their use should be limited solely to embolization at the site of rupture of an aneurysm, because they may not only give rise to the formation of collateral circuits to the tumor, but also cause revascularization of the metallic coil and pose a hindrance at the next intervention.Our present patient has been under periodic follow-up for over 5 years; a long-sustained contraction of the hematoma and angiomyomatous components of the tumor has been evident from the time the treatment was administered until date. Although introduction of dialysis may hardly be avoidable with possible enlargement of the AML arising from the left kidney in the future , its avoSelective embolization is recommended even in cases with rupture of a giant AML developing in a unilateral residual kidney because embolization of the tumor vasculature and the attempt to conserve normal renal parenchyma to the greatest extent possible provides not only hemostasis, but also results in reduction of the tumor diameter and conservation of renal function. In the present case, the adoption of this therapeutic strategy with concomitant CT during selective arteriography yielded favorable outcomes both in the short-term and in the long-term.


Inflammatory myofibroblastic tumour is a distinctive mesenchymal neoplasm, composed of a variable admixture of myofibroblastic spindle-shaped and inflammatory cells which were originally described in the lung, as a nonneoplastic lesion and designated as an inflammatory pseudotumour. The lack of certainty of the IMTs pathogenesis is reflected in the large number of terms which have been attributed to this lesion. Recent genetic and molecular studies of IMTs have showed chromosomal abnormalities of 2p23 resulting in a rearrangement of the anaplasic lymphoma kinase gene and have also provided evidence for a monoclonal, noeplastic origin for IMT.Occurrence of IMT in the kidney is very rare, and to our knowledge, only 30 such cases have been described in the literature.This report describes an original case of an ALK positive IMT of the kidney associated with renal pelvic calculus which we believe has never been reported. The differential diagnosis of IMTs will also be discussed. The inflammatory myofibroblastic tumours (IMT) correspond to a distinct organic entity, made up of spindle-shaped cells myofibroblastic frays with an inflammatory infiltrate formed by lymphocytes, plasmocytes and eosinophilic cells. These lesions are ubiquitous, preferentially interesting the lung, the mésentery and the epiploon . When itWe report a new case of inflammatory myofibroblastic kidney tumour, associated with a renal pelvis lithiasis. In the meantime, we will insist on the pathogenesis discussion of these tumours and on their anatomopathologic diagnosis.A 35-year-old Moroccan man, without particular pathological history, was admitted as he was suffering from a one-year recurrent renal colic. The clinical examination of the patient revealed a big left kidney without the other associated clinical anomalies.The biological investigation did not reveal any particular anomaly. Radiological exploration was also carried out, by the means of a renal ultrasonography and an abdominal scanner. It has unveiled a tumor measuring 5 cm of main axe, located in the lower pole of the left kidney, with an extension to the hilum. A surgery with total nephrectomy was performed on the patient in an effort to deal with this unusual renal tumour. The macroscopic examination showed a part of left total nephrectomy of 10 × 8 × 6 cm. With the opening, it contained a heterogeneous lesion of a tumoral aspect, measuring 5 × 4 × 4 cm, with blurry boundaries and occupying the lower pole of the kidney. This tumor was extended to the hilum and had no recognizable vascular structure. There was no crossing of the renal capsule. Histologically, the tumor was formed by a proliferation of spindle-shaped cells, of myofibroblastic look  mixed wiThe morphological and phenotypical features, reported in our case, are those of an inflammatory myofibroblastic tumour (IMT), characterized by spindle-shaped cells and myofibroblastic proliferation, and associated with a polymorphic inflammatory infiltration, particularly rich in plasmocytic cells which are contained within an oedematous stroma. The pathogenesis of the IMT has been for a long time in the center of a debate whether their nature is reactive or tumoral. Initially, they were described in the lung, under the post-inflammatory tumour term . Later oRecent studies show that a part of these lesions, at the very least, constitutes a true entity of neoplasic nature. Indeed, Griffin et al. carried out a study on 3 new cases and of the retrospective analysis of the caryotypes previously published. It revealed a recurring and non-random anomaly of the chromosomal band 2p23 that is located on the distal segment of the court arm of chromosome 2 with a point of break in the ALK gene (Anaplastic Lymphoma Kinase), coding for a receiver of tyrosin kinase and already known for its implication in the anaplasic lymphoma with big cells [Therefore this analysis confirmed dismissed the reactive nature of the IMT, and confirmed they were clonal and tumoral in connection with the deterioration of the ALK gene along with the existence of various genes playing a role in this fusion.Cases of association of IMT with some bacterial or viral infections such as HHV-8 virus (human herpes virus-8) were reported in the literature . In the The first one is the existence of a cause-effect relationship between the tumour and the lithiasis as the tumour can support the constitution of the lithiasis by obstructing the urinary excretion. In addition, a lithiasis could be at the origin of an excessive reactive inflammatory process, carrying out a pseudotumoral aspect, however, this type of lesion gives a negative immunolabelling with the ALK, but the exact opposite is detected in our patient .The second one assumption is the fortuitous association which seems more probable in the case of our patient.The IMT of the kidney, often asymptomatic, can be revealed by lumbagos, hématuria, hydronephrosis, nonspecific systemic demonstrations such as prolonged hyperthermia or by the means of biological inflammatory syndromes . DespiteAccording to the morphological profile of the IMT, the differential diagnosis must include various types of tumours with spindle-shaped kidney cells, but also reactive processes. Indeed, when the myofibroblastic proliferation develops within an oedématous stroma and/or myxoi, the aspect can recall a visceral nodular fasciite. Nevertheless, the IMT contains traditionally the inflammatory infiltrates that is rich in plasmocytes, which are often missing in the nodular facciite . In the The IMT constitute a distinct tumoral entity, with a genetic reorganization of ALK with the participation of a variety of genes in the fusion and the activation of detectable ALK in immunohistochemistry. The unusual association with a lithiasis of the renal pelvis, described is probably a coincidence. The extremely rare localization of the IMT in the bladder leads to the morphological differential diagnosis with several kidney tumours with spindle-shaped cells and a visceral nodular fasciite.


Gallbladder torsion is an uncommon clinical entity and a difficult condition to diagnose preoperatively. Since its first description in 1898 by Wendel there have been over 500 documented cases in the literature. It is known to occur when there is rotation of the gallbladder along the axis of the cystic duct and vascular pedicle. Except for isolated cases reported in childhood, this disease is more frequently encountered in the elderly with 85% of the cases reported between the ages of 60 and 80 years. There is a female preponderance with a female to male ratio of 3:1. Gallbladder torsion typically presents as an acute abdomen requiring emergency surgery but preoperative diagnosis of gallbladder torsion is difficult and most cases are found as a surprise at surgery.We report a case of acute gallbladder torsion in an elderly lady and review the clinical aspect of the disease. Gallbladder torsion is an uncommon clinical entity and a difficult condition to diagnose preoperatively. It is known to occur when there is rotation of the gallbladder along the axis of the cystic duct and vascular pedicle. Since its first description in 1898 by Wendel, there have been over 500 documented cases in the literature . Except The common causative factor is an abnormality in the anatomy of the gallbladder and its associated vascular pedicle. In general, this is mainly due to the two mesenteric anatomical variations including either a wide or a long mesentery with cystic artery and duct. This is referred to as a 'floating' or 'pedunculated' gallbladder ,3,5-7. IWe report a case of acute gallbladder torsion in an elderly lady and review the clinical aspects of the disease.A 79 year old lady presented to Accident & Emergency department with a two day history of sudden onset colicky abdominal pain associated with vomiting and progressive abdominal distension. She had failed to pass any faeces or flatus 24 hours prior to presentation. She denied any history of recent change in bowel habit or weight loss. There was no significant relevant past medical history.3 25.1 & BE +2.3. Laboratory blood tests revealed mildly deranged liver function tests (LFT) , C-reactive protein (CRP) 152 mg/L and white blood cell (WBC) count 20 × 109 (neutrophils 17.8 × 109). She was admitted with the provisional diagnosis of acute large bowel obstruction secondary to a possible transverse colon tumour. Differential diagnoses of acute cholecystitis and mesenteric cyst torsion was entertained.She was dehydrated at presentation with the following vital signs: HR-107 b/min, BP-119/50 mmHg, T-36.5°C, oxygen saturation-97% on air. Abdomen was distended and resonant to percussion with a palpable tender central abdominal mass around the umbilicus measuring 5 cm by 6 cm. Bowel sounds and digital rectal examinations were normal. Erect chest X-ray was unremarkable, whilst abdominal X-ray showed normal small bowel gas pattern and right sided colonic faecal loading. Arterial blood gas on air showed pH 7.49, HCOShe was resuscitated with IV fluids and an emergency computerized tomography (CT) scan of the abdomen and pelvis was performed. This was reported as showing an oval cystic lesion in the central abdomen, with an AP diameter of 6 cm and a transverse diameter of 9 cm. This structure was unilocular with no connection to the bowel or mesentery but compressing the transverse colon Figures  &2. Therth day post surgery. The delay in discharge was due to social reason.Emergency laparotomy through a midline incision was performed and findings at operation included a large, gangrenous gallbladder with a 360 degree clockwise torsion on its long pedicle. The gallbladder was distinctly suspended free from the liver edge and lying over the greater omentum and transverse colon with some haemorrhagic peritoneal fluid reaction. The torted pedicle was undone and cholecystectomy was performed. The opened gallbladder had complete hemorrhagic infarct with wall thickness of about 1 cm across Figures . The patTorsion of the gallbladder is a rare clinical condition of the hepatobiliary system, with a reported clinical incidence of 1 in 365,520 hospital admissions ,9. In faThe aetiology of gallbladder torsion is unknown; however, there are several factors that have been postulated as playing significant causative roles. There are two requirements for developing gallbladder torsion, the presence of a long mesentery or a very short/absent mesentery allowing mobility of the gallbladder along its vertical axis and the other factor is generalized loss of elastic tissue and visceroptosis in the elderly. These factors contribute to non-fixation of the gallbladder to the inferior margin of the liver causing the gallbladder to hang freely in the peritoneal cavity ,3,5-8,10The symptoms of gallbladder torsion are largely non-specific and this makes preoperative diagnosis difficult on the basis of history and physical examinations alone ,9. PatieLaboratory investigations are largely unhelpful. Raised white blood cell count and C-reactive protein are frequent findings especially with the onset of gangrenous cholecystitis. Liver function tests are usually normal in patients with gallbladder torsion . ImagingKitagawa H et al  providedEarly diagnosis of gallbladder torsion is important in order to avoid the complication of perforation and the attendant bilious peritonitis. Early diagnosis and prompt surgical interventions have been shown to reduce the mortality to less than 5% . EmergenGallbladder torsion is rare and therefore requires a high index of suspicion for early preoperative diagnosis and prompt surgical intervention. This diagnosis should be considered in the setting of an elderly woman with atypical or non-resolving symptoms and signs of acute cholecystitis in spite of the use of adequate antibiotic therapy. Increasing incidence of gallbladder torsion is being encountered today and this is probably due to unreserved use of imaging investigations and laparoscopy. Early diagnostic imaging investigations and prompt cholecystectomy is the aim in order to achieve best patient outcome.The author(s) declare that they have no competing interests.GJ participated in the admission and the care of this patient, the conception, design, data collection and interpretation, manuscript preparation and literature search. AAA participated in the admission and the care of this patient, the conception, design, data collection and interpretation, manuscript preparation and literature search. HH participated in the admission and the care of this patient, the conception, design, data collection and interpretation, manuscript preparation and literature search. All authors read and approved the final manuscript.


Low-grade papillary adenocarcinomas of the sinonasal tract are rare neoplasms. Over recent years, little doubt remains that this tumour represents a separate entity based on morphology, ultrastructural features and behaviour. We outline a case of this rare entity displaying a not hitherto described immunophenotype.A 32 year old man presented recurrent epistaxis was evaluated with endoscopy which revealed a well circumscribed pedunculated mass lesion in left nares. The mass was arising from the nasal septum which was excised along with the mass. The biopsy revealed low-grade, non-intestinal type sinonasal tubulopapillary adenocarcinoma.TTF-1 immunoreactivity in absence of thyroid or pulmonary primary in the present case remains an enigma. However, this raises the possibility of the utility of this antibody to predict a better clinical outcome in the subset of low grade non-intestinal sinonasal adenocarcinoma. More cases of similar morphological appearance may need to be examined for TTF-1 immunoreactivity and clinically followed up to establish this theory. Sinonasal adenocarcinomas are rare tumours accounting for 0.4%  of all hA 32 year old man who had recurrent episodes of epistaxis was seen in the ENT outpatient clinic. Flexible endoscopy revealed deviation of the nasal septum to the left. Arising from the posterior end of the left nasal septum was a pedunculated well-circumscribed lesion. Magnetic resonance imaging revealed no other abnormalities. At operation, a lobulated solid mass was seen. The mucosa anterior to the mass had become detached. The underlying bone was removed but did not look involved. Postoperative recovery was uneventful and he was discharged the next day. The lesion was suspected to be a haemangioma. Previous episodes of epistaxis were treated with silver nitrate cautery. The patient has no significant past medical history. He is a non-smoker, was not on any regular medication and had no relevant occupational history. Subsequently, the patient had two further operations. Firstly, removal of the posterior aspect of the nasal septum was performed four months after removal of this mass. Secondly, a biopsy of the nostril was undertaken. The former revealed mucosal fragments incorporating seromucinous glands with intervening chronic inflammation of the stroma but no evidence of residual adenocarcinoma. The latter showed inflammatory granulation tissue around suture granulomata from previous surgery. Since initial presentation over two years ago, the patient remains free of recurrence or metastatic disease and does not have any lesions in his lungs or thyroid gland.Macroscopically, two yellow-white polypoid fragments of tissue, measuring 10 and 4 mm in maximum dimension were received. Histologically, these fragments were partly covered by focally ulcerated squamous epithelium. The underlying stroma was infiltrated by a neoplasm with a complex papillary and tubular configuration, lined by moderately dysplastic pale columnar epithelium with intervening spindle shaped cellsFigure .Immunohistochemical labelling revealed diffuse positivity with antibodies to EMA, CAM 5.2, CK 7, CK 19 and TTF-1 Figure . The celAs described recently , low-graet al. [The importance of recognition and separation of this neoplasm from other types of sinonasal adenocarcinoma is critical as it virtually never metastasizes and has an excellent prognosis. Unlike this case, Franchi et al. , have reThere is no published data on the role of TTF-1 in adult primary nasal adenocarcinomas. To date, we are unaware of any occult thyroid or pulmonary tumours in our patient to explain the TTF-1 immunoreactivity. The significance of this unexpected immunohistochemical labelling remains an enigma. However, this unusual TTF-1 positivity raises the possibility of the utility of this antibody to predict a better clinical outcome in the subset of low grade non-intestinal sinonasal adenocarcinoma. More cases of similar morphological appearance may need to be examined for TTF-1 immunoreactivity and clinically followed up to establish this theory.The authors declare that they have no competing interests.AB conducted a literature search, took the photomicrographs and drafted the manuscript; KEP edited the manuscript; KPG is the consultant who reported the biopsies and proofread the final manuscript. All authors read and approved the final manuscript.


Intussusception is a pediatric condition that rarely presents in adults. In this article, we report a case of a 36 year-old man initially presenting with abdominal pain and rectal prolapse, however, surgical reduction of the rectal prolapse did no relief his symptoms. Physical examination, abdominal plain film, barium enema and colonoscopy confirmed the presence of a large intra-abdominal mass, but the origin of the mass was revealed only upon laparotomy. During the surgery, it was noted that the ileum and the sigmoid colon was connected by a 15 cm × 12cm mass, covered by an extremely dilated intestinal tissue. The resected tissue pathology demonstrated a 9 cm × 6 cm × 5 cm submucosal lipoma at the ileocecal junction without evidence of malignancy. The patient's post-surgical course was uneventful. Diagnostic and therapeutic problems related to adult intussusception are reviewed. Intussusception is the telescoping of one segment of the gastrointestinal tract into an adjacent one. It is usually known as a pediatric condition, found in adults with a low frequency of about 2–3 per 100,000. As the condition is relatively rare, it is often not considered in the differential diagnosis of adult patients with vague abdominal complaints. Moreover, the triad of symptoms: cramping, vomiting, and rectal bleeding are not as obvious in adults as among children, thus, making it difficult to diagnose with an even greater delay before treatment. However, as opposed to children, 90% of adult intussusceptions are associated with an identifiable etiology. About two-thirds are due to malignant tumors and with less than one-third from benign processes. Hence, there is definitely a need to identify the underlying causes of adult intussusception and provide the treatment required.A 36 year-old man presented to the local hospital with the chief complaint of abdominal pain and "something protruded from the rectum." He had a two-month history of diarrhoea but now had constipation for the past 2 weeks. The prolapse happened that afternoon when he was straining to defecate. Physical examination was unremarkable except for a 15 cm × 15 cm, firm, immobile, non-tender mass in the left lower quadrant of the abdomen. On rectal examination, the prolapsed was an irregular soft mass non-contiguous with the anus. An immediate surgical reduction for prolapse was performed; however, it did not relieve the abdominal pain. A subsequent barium enema  telescopes into an adjacent distal segment (intussuscipiens). The symppearance,5. OtherIn this case, the patient was initially diagnosed with rectal prolapse, which should not interfere in the diagnosis of additional intussusception. In the event of a rectal prolapse, continuity may be detected through palpation between the perianal/anal tissue and the protruding tissue. In contrast, in intussusception no palpable continuity may be felt. The barium enema of the patient provided some clues to the diagnosis of intussusception such as the irregular filling defect around the large mass and the sharp end of the proximal colon, the irregular shadow in the lumen between the sigmoid colon and the rectum, which might be the prolapsed intestine. However, they were neglected. Instead, a colonoscopy instead of computed tomography was performed at the local hospital. Though this led to a number of findings important for the final diagnosis, it is still questionable whether this was a worthwhile procedure. Furthermore, the biopsy during the colonoscopy appeared unnecessary, as this patient needed a laparotomy due to the large mass occluding the colon. Though we had not performed a CT scanning before the surgery, we doubted its significance in helping with the diagnosis or surgery as the intussusception of the patient seemed to be too complicated.After several days of delay, he finally underwent laparotomy. On opening of the abdomen, we were astonished by what we saw. The left abdominal quadrants were occupied by a large mass covered with an extremely distended intestinal tissue Fig . The intet al, about 80–90% of intussusceptions in adults are secondary to an underlying pathology, with approximately 65% due to benign or malignant neoplasms[Early reports advocate reduction of adult intussusception instead of resection. Howeverneoplasms. HoweverIn this case, after confirming benign lesion and the possible reduction, our group performed a manual reduction followed by a subtotal colectomy, allowing more ileum and sigmoid colon to be preserved. We decided that the prognostic factor involved the underlying nature of the lesion and the remaining length of functional intestine. Thus, the decision on reduction versus resection should be considered on the basis of survival and future quality of life.Adult intussusception occurs infrequently and differs from pediatric intussusception in its presentation, etiology, and treatment. Diagnosis can be delayed because of its long-lasting, intermittent, and non-specific symptoms. A more frequent use of computed tomography in the evaluation of patients with uncharacteristic abdominal pain may allow the condition to be more reliably diagnosed. Whether resection or reduction of the bowel tissue involved is still controversial. However, many speculate against reduction before resection, especially when taking into account cases where the bowel is nonviable or when malignancy is suspected. Total colectomy is not recommended if the character of the mass is not defined. Finally, exploratory laparotomy, though not the first choice for patients whose etiology remains unknown, is a necessary and sometimes effective solution.Common symptoms such as abdominal pain, palpable masses should not be neglected in the initial assessment of patients.• Adult intussusception, though rare, should be one of the differential diagnoses of vague abdominal pain, especially when found concomitantly with a palpable mass in the abdomen.• Computed tomography is the cornerstone for the diagnosis of intussusception.• The underlying nature of the lesion leading to the intussusception and the remnant length of functional intestine should be taken into account when one is considering treatment of adult intussusception.• The authors declare that they have no competing interests.RC analyzed and interpreted the patient data regarding the overall condition of the patient, and was a major contributor in writing the manuscript. HZ performed the surgery, and was also a major contributor in writing the manuscript. XS supervised the surgery and gave helpful suggestions on the surgery. YM and XL attended the surgery and contributed a lot to the writing and reviewing the manuscript. YY helped with reviewing the manuscript. All authors read and approved the final manuscript.Written informed consent was obtained from the patient and any accompanying images. A copy of the consent is available for review by the Editor-in-Chief of this journal.


Failure of total hip arthroplasty through septic or aseptic loosening, periprosthetic fracture, or recurrent dislocation is well recognized and understood. We present an unusual cause of failure of total hip replacement which occurred on a 79 year old gentleman: that of prosthetic loosening secondary to malignant infiltration around components. Our aim is to highlight the fact that malignant infiltration should be considered as part of the differential diagnosis in aseptic and septic loosening of prosthetic implants. A 79-year-old Caucasian gentleman, with no history of any malignancy, underwent a left cemented total hip replacement (THR) for osteoarthritis and was symptom free post-operatively Figure . At 6 moThe patient was then referred to the oncologist for palliative treatment and died 6 months later.Metastatic spread to a joint replacement is exceptional. To date, there have been very few reports in the literature documenting periprosthetic metastatic disease as a mode of failure in total hip and knee arthroplasty. These consisted of a non-Hodgkin lymphoma , an immuA metastatic lesion in a patient who has undergone prior THR may appear as solitary periprosthetic lucency and may be misinterpreted as aseptic periprosthetic osteolysis. Aseptic osteolysis may be extensive in size, appears as endosteal scalloping and does not usually invade the outer cortex. Single lesions that involve the whole cortex, appear soon after implantation, are painful and progress rapidly are not typical of aseptic osteolysis and should raise a suspicion of malignancy.Furthermore, according to study done by Mohler et al , early lOne should maintain a high index of suspicion and consider metastatic disease as a differential diagnosis in cases of aseptic loosening, particularly when there is rapid progression of symptoms, the history is atypical, the patient has a history of malignant disease and the osteolytic lesion involves the outer cortex. If these features are present, the lesion should be biopsied and appropriate radiological and haematological investigations should be considered."Written informed consent was obtained from the patients' next of kin 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."The authors declare that they have no competing interests.AD and ASD wrote the draft of the manuscript and performed the literature search; TNB revised the manuscript for intellectual content; JRP and WEAH performed the surgical procedure.