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1.  Parainfectious encephalomyeloradiculitis associated with bacterial meningitis: a case report 
Acute disseminated encephalomyelitis in adulthood occurs in most cases after a viral infection. Acute disseminated encephalomyelitis associated with bacterial meningitis, however, is quite rare.
Case presentation
An 82-year-old Japanese woman presented with a fever and somnolence. Increased neutrophil count and protein content, and decreased glucose levels in her cerebrospinal fluid initially suggested bacterial meningitis. Brain magnetic resonance imaging on admission showed bilateral symmetrical lesions in her brainstem and her cerebellum. She was diagnosed with acute disseminated encephalomyelitis following bacterial meningitis. Even though appropriate antibiotic and steroid treatment improved her symptoms, she developed transverse myelitis and lumbosacral polyradiculitis on day 9.
Parainfectious encephalomyeloradiculitis, a variant of acute disseminated encephalomyelitis, is a unique neurological syndrome that may be caused by bacterial infection in the central nervous system.
PMCID: PMC4332421
Acute disseminated encephalomyelitis; Bacterial meningitis; Encephalomyeloradiculitis; Polyradiculoneuropathy; Post-infectious
2.  Chylous ascites and lymphangiectasia in focal segmental glomerulosclerosis – a rare coexistence: a case report 
Nephrotic syndrome is considered a rare cause of chylous ascites. Intestinal lymphangiectasia in a background of chylous ascites and without any lymphatic obstruction has been reported in association with yellow nail syndrome, which is a rare clinical occurrence in itself. The existence of chylous ascites, duodenal and splenic lymphangiectasia (without any lymphatic obstruction) and nephrotic syndrome in the form of focal segmental glomerulosclerosis in the same patient makes this case the first of its kind to be reported in the literature.
Case presentation
Here we report the case of a 54-year-old Asian man who presented with recurrent episodes of anasarca for approximately 25 years. He was subsequently found to have chylous ascites, lymphangiectasia and persistent proteinuria. A renal biopsy revealed focal segmental glomerulosclerosis, not otherwise specified. A lymphangiogram, which was performed with the purpose of addressing the intestinal lymphangiectasia, failed to demonstrate any abnormality of lymphatic channels. He was put on oral steroids with consequent remission of his oedema and proteinuria.
This case highlights the fact that duodenal and splenic lymphangiectasia can exist in a scenario of chylous ascites without any obvious obstruction of lymphatic channels and in the absence of yellow nail syndrome. This case also signifies that chylous ascites may be a rare presenting feature of nephrotic syndrome and hence this aspect should be considered while in diagnostic dilemma regarding such a clinical presentation.
PMCID: PMC4333874
Chylous ascites; Focal segmental glomerulosclerosis; Lymphangiectasia
3.  Successful en bloc resection of recurrent hepatocellular carcinoma directly invading the abdominal wall: a case report 
Repeat hepatic resection has previously been reported as the most effective treatment for recurrence of intrahepatic carcinoma. To the best of our knowledge, en bloc resection of recurrent hepatocellular carcinoma directly invading the abdominal wall has not been previously reported.
Case presentation
In September 2012, a 64-year-old Chinese male patient was referred to our hospital because of primary hepatocellular carcinoma located in Couinaud’s segments III and V. Our patient first had a hepatectomy of the liver. Ten months later, he presented with an abdominal wall mass and upper abdominal pain. Computed tomography and magnetic resonance imaging scans demonstrated a 10cm tumor in his left liver with extrahepatic metastases in his abdominal wall. It was determined that he had recurrent hepatocellular carcinoma associated with direct invasion into his abdominal wall. He had an en bloc left hepatectomy with resection of the tumor in his abdominal wall. A pathological examination of the resected specimen confirmed the diagnosis of hepatocellular carcinoma involving the abdominal wall. Disease-free margins of resection were achieved. Our patient’s postoperative course was uneventful. Eight months after the last surgery, our patient died owing to recurrence and distal metastasis.
Direct invasion of hepatocellular carcinoma into the abdominal wall is rarely encountered. Complete surgical resection should be considered in patients with an appropriate hepatic functional reserve, with consideration of the technical difficulty relating to tumor involvement with surrounding tissues.
PMCID: PMC4333836  PMID: 25609080
Hepatectomy; Hepatocellular carcinoma; Recurrence
4.  Set screw fracture with cage dislocation after two-level transforaminal lumbar interbody fusion (TLIF): a case report 
Transforaminal lumbar interbody fusion is a popular procedure used to achieve spondylodesis in patients with degenerative lumbar spinal diseases. We present a rare case of a patient with a set screw fracture with cage dislocation after an open transforaminal lumbar interbody fusion procedure. To the best of our knowledge, this case is the first of its kind to be reported.
Case presentation
A 44-year-old Caucasian woman attended a follow-up appointment at our hospital 3 months after treatment for second-degree lumbar spondylolisthesis (L4/L5) and osteochondrosis (L5/S1) with transforaminal lumbar interbody fusion and dorsal spondylodesis. She complained of severe leg pain on the left side. Her physical examination revealed a normal neurological status, except for paresthesia of the entire left lower limb and at the ball of the left foot. Radiological imaging showed breaking of the set screws with cage dislocation. Surgical revision was then performed with exchange of the whole dorsal instrumentation and the dislocated cage. Six weeks post-operatively, the patient was seen again at our clinic without neurological complaints, except for decreased sensitivity on the dorsum of her left foot. The wound healing and radiological follow-up were uneventful.
Hardware-related complications are rarely seen in patients with open transforaminal lumbar interbody fusion, but must be kept in mind and can potentially cause severe neurological deficits.
PMCID: PMC4333885  PMID: 25609204
Complication; Fracture; Set screw; TLIF; Transforaminal lumbar interbody fusion
5.  Giant anal condyloma (giant condyloma acuminatum of anus) after allogeneic bone marrow transplantation associated with human papillomavirus: a case report 
Condyloma acuminatum are caused by human papillomavirus. Giant condyloma acuminatum is a locally invasive, destructive, and large sized mass. Risk factors for the development of giant condyloma acuminatum include an immunodeficient state, such as human immunodeficiency virus infection, post-organ transplantation, or post-allogeneic bone marrow transplantation. However, reports of giant condyloma after bone marrow transplantation are extremely rare (0.3 to 1.3%). The standard treatment for giant condyloma acuminatum is recommended as wide surgical resection due to its high rate of success and low rate of recurrence.
Case presentation
A 31-year-old Korean man presented to our hospital with anal discomfort for more than one month due to a protruding mass. He had a history of BCR-ABL-positive acute lymphoblastic leukemia and had undergone an allogenic stem cell transplantation. Gross findings revealed a large perianal cauliflower-like mass over 7cm in size with invasion of the anal orifice. He was diagnosed with giant anal condyloma occurring after an allogeneic bone marrow transplantation. However, we achieved successful treatment using a combination of topical podophyllin and cryotherapy and transanal surgical excision, followed by bleomycin irrigation.
We report an extremely rare case of giant condyloma acuminatum of anus due to human papillomavirus type six in a patient with acute lymphoblastic leukemia following an allogeneic bone marrow transplantation. The tumor was successfully treated with a combination of topical podophyllin and cryotherapy and transanal surgical excision, followed by bleomycin irrigation.
PMCID: PMC4334849  PMID: 25597932
Condyloma; HPV; Allogenic bone marrow transplantation; Anus condyloma
6.  Surgical treatment of scoliosis in Rubinstein-Taybi syndrome type 2: a case report 
Rubinstein-Taybi syndrome is an autosomal dominant disorder resulting in congenital craniofacial deformities, and divided into types 1 and 2. Scoliosis has not been reported as one of the extra-cranial manifestations of Rubinstein-Taybi syndrome type 2.
Case presentation
We present a 14-year-old British Caucasian girl with Rubinstein-Taybi type 2 syndrome who developed a severe double thoracic scoliosis measuring 39° and 68° respectively. Her scoliosis was associated with thoracic hypokyphosis, causing a marked reduction in the anteroposterior diameter of her chest and consequent severe restrictive lung disease. The deformity was noted by her local pediatrician as part of a chest infection assessment when she was aged 13 years, and gradually progressed as the result of spinal growth. Our patient underwent a posterior spinal arthrodesis using a single concave pedicle hook and screw rod construct and locally harvested autologous graft supplemented by allograft bone. This spinal fixation technique was selected because of our patient’s low body weight to avoid prominence of the instrumentation causing skin healing problems and pain. Her scoliosis was corrected to 18° and 30° and we achieved a balanced spine in the coronal and sagittal planes. An underarm spinal jacket was provided for six months after surgery. During her latest follow-up at skeletal maturity, our patient had an excellent cosmetic outcome with no loss of deformity correction or detected pseudoarthrosis and a normal level of activities.
Scoliosis can develop in young children with Rubinstein-Taybi syndrome type 2, with the deformity deteriorating around the pubertal growth spurt. Surgical treatment can correct the deformity, balance the spine and prevent mechanical back pain. It can also stabilize the chest area and avoid respiratory complications developing as the scoliosis progresses, which can result in severe restrictive pulmonary disease. The use of single concave instrumentation is indicated in very slim patients with poor muscle bulk; in our patient, this produced satisfactory deformity correction and a favorable outcome at completion of growth. Peri-operative care in this group of patients can be very challenging because of associated co-morbidities as well as the presence of severe behavioral issues that result in poor patient compliance.
PMCID: PMC4334754  PMID: 25596810
Posterior spinal fusion; Rubinstein-Taybi syndrome; Scoliosis; Surgical treatment
7.  A 42-year-old patient presenting with femoral head migration after hemiarthroplasty performed 22 years earlier: a case report 
Treatment of femoral neck fractures in young adults may require total hip arthroplasty or hip hemiarthroplasty using a bipolar cup. The latter can, however, result in migration of the femoral head and poor long-term results.
Case presentation
We report a case of femoral head migration after hemiarthroplasty performed for femoral neck fracture that had occurred 22 years earlier, when the patient (a Japanese man) was 20 years old. He experienced peri-prosthetic fracture of the femur, subsequent migration of the prosthesis, and a massive bone defect of the pelvic side acetabular roof. After bone union of the femoral shaft fracture, the patient was referred to our hospital for reconstruction of the acetabular roof. Intra-operatively, we placed two alloimplants of bone from around the transplanted femoral head into the weight-bearing region of the acetabular roof using an impaction bone graft method. We then implanted an acetabular roof reinforcement plate and a cemented polyethylene cup in the position of the original acetabular cup. Eighteen months post-operatively, X-rays showed union of the transplanted bone.
Treatment of femoral neck fractures in young adults is usually accomplished by osteosynthesis, but it may be complicated by femoral head avascular necrosis or by infection or osteomyelitis. In such cases, once an infection has subsided, either hip hemiarthroplasty using a bipolar cup or total hip arthroplasty may be required. However, if the acetabular side articular cartilage is damaged, a bipolar cup should not be used. Total hip arthroplasty should be performed to prevent migration of the implant.
PMCID: PMC4326395  PMID: 25592554
Acetabular articular cartilage; Femoral head migration; Femoral neck fracture; Hemiarthroplasty; Infection
8.  A case of type B lactic acidosis as a complication of chronic myelomonocytic leukaemia: a case report and review of the literature 
Type B lactic acidosis represents a rare and often lethal complication of haematological malignancy. Here, we present a patient who developed a type B lactic acidosis presumably due to a concurrent chronic myelomonocytic leukaemia. Upon swift initiation of cytoreductive chemotherapy (doxorubicin), the lactic acidosis was rapidly brought under control. This case adds to the literature reporting other haematological malignancies that can cause a type B lactic acidosis and its successful treatment.
Case presentation
We report the case of a 77-year-old Caucasian man brought to our Accident and Emergency department following an unwitnessed collapse; he was found surrounded by coffee-ground vomit. Although haemodynamically stable on admission, he rapidly deteriorated as his lactic acid rose. An initial arterial blood gas revealed a pH of 7.27 and lactate of 18mmol/L (peaking at 21mmol/L).
A high degree of clinical suspicion for haematological malignancy should be held when presented with a patient with lactic acidosis in clinical practice, even without evidence of poor oxygenation or another cause. Treatment with emergency chemotherapy, in lieu of a definitive diagnosis, was rapidly successful at lowering lactate levels within 8 hours. This may suggest a causal and perhaps direct relationship between lactic acid production and the presence of leukemic cells. Veno-venous haemofiltration had no apparent effect on reducing the lactic acidosis and therefore its benefit is questioned in this setting, especially at the cost of delaying chemotherapy. In the face of a life-threatening lactic acidosis, pragmatic clinical judgement alone may justify the rapid initiation of chemotherapy.
PMCID: PMC4325955
Acidosis; Chronic myelomonocytic leukaemia; Haematological malignancy; Type B lactic acidosis
9.  Elderly onset intramedullary epidermoid cyst in the conus medullaris: a case report 
Epidermoid cysts are known as embryonic or acquired ectopic aberrations of the ectoderm. To the best of our knowledge, there are only a few reports of elderly onset intramedullary epidermoid cysts. We report a case of elderly onset intramedullary epidermoid cyst at the conus medullaris.
Case presentation
A 63-year-old Japanese woman working as a farmer presented with slowly progressive gait disturbance and voiding dysfunction. A magnetic resonance imaging scan revealed an intramedullary mass lesion at L1 to L3. We diagnosed the lesion as an intramedullary spinal cord tumor. A laminectomy was performed at the level of Th12 to L3. Upon spinal cord dissection, a yellowish milky exudation erupted from the cystic lesion. We resected white cartilage-like pieces from the cystic cavity. Because the wall of the cystic lesion tightly adhered to the spinal cord parenchyma, we abandoned complete resection of the cyst wall. The pathological diagnosis was an epidermoid cyst.
We propose that evacuation of the cyst contents is preferable, especially in cases with elderly onset and congenital origin.
Electronic supplementary material
The online version of this article (doi:10.1186/1752-1947-9-7) contains supplementary material, which is available to authorized users.
PMCID: PMC4326493  PMID: 25582755
Epidermoid cyst; Intramedullary tumor; Conus medullaris
10.  Urgent endovascular ligature of a ruptured splenic artery pseudoaneurysm in a patient with acute pancreatitis: a case report 
We report on the successful endovascular treatment of a ruptured splenic artery pseudoaneurysm. Our patient had acute pancreatitis superimposed on chronic calcific pancreatitis and chronic renal impairment. Contrast-enhanced ultrasonography was used to assess post-embolization results.
Case presentation
Our patient was a 67-year-old white Caucasian man with recurrent pancreatitis. Computed tomography angiography showed a pancreatic pseudocyst with a ruptured pseudoaneurysm, which was successfully embolized using an endovascular percutaneous approach. At six months, persistent renal failure led to contrast-enhanced ultrasonography. This confirmed the absence of turbulent blood flow and extravasation of contrast medium in the pseudocyst.
Our experience with this case leads us to support the role of interventional radiology as a first-line treatment tool. Contrast-enhanced ultrasonography can be used to follow-up embolization procedures in patients with impaired renal function.
PMCID: PMC4326335  PMID: 25575735
Acute pancreatitis; Contrast-enhanced ultrasonography; Endovascular ligature; Impaired renal function; Pancreatic pseudoaneurysms
11.  Association of laryngeal and nasopharyngeal tuberculosis: a case report 
To the best of our knowledge, the association of nasopharyngeal and laryngeal tuberculosis has never been described before in the literature. We report here a first observation.
Case presentation
We report the case of a 38-year-old Arab man who presented with an isolated hoarseness. Radiological and endoscopic examinations showed a thickening of the left lateral wall of his nasopharynx and the left vocal cord. Pathology revealed the diagnosis of tuberculosis of both localizations. He received a 6-month antituberculous chemotherapy with a satisfying uneventful evolution.
Tuberculosis should be considered in the differential diagnosis of soft tissue masses of the head and neck, particularly when the imaging findings and clinical presentation are atypical. The diagnosis of tuberculosis is mainly based on histopathological and/or bacteriological examination.
PMCID: PMC4326367  PMID: 25560475
Larynx; Nasopharynx; Tuberculosis
12.  Persistent parvovirus B19 viremia with chronic arthralgia treated with ascorbic acid: a case report 
According to some studies, ascorbic acid possesses antiviral properties. These studies were mainly focused on the common cold, with very few focusing on other viral infections.
Case presentation
We report the case of a 54-year-old Caucasian woman with chronic arthralgia due to persistent parvovirus B19 viremia. High doses of ascorbic acid treatment were initiated due to the failure of conventional analgesic therapy. Clinical benefit was observed with a simultaneous loss of biological parvovirus B19 viremia.
This observation shows a potential benefit of the use of ascorbic acid against parvovirus B19 infections, even if this case is not sufficient to draw any definite conclusions.
PMCID: PMC4326192
Ascorbic acid; Parvovirus B19; Arthralgia; Chronic
13.  Intraparenchymal meningioma mimicking cavernous malformation: a case report and review of the literature 
A primary intraparenchymal meningioma located in the subcortical region of the brain without a dural attachment is extremely rare. To the best of our knowledge, this is the first report showing that meningioma can mimic cavernous malformations.
Case presentation
We present the case of a 42-year-old German man who presented to our institution with seizure. Both computed tomography and magnetic resonance imaging scans showed characters of an intra-axial subcortical lesion with a ‘popcorn’ appearance and hemosiderin deposits in the right parietal lobe. The initial diagnosis was cavernous malformation. Intraoperatively, the lesion presented as a subcortical mass that had no connection to the dura or the ventricle. The histological diagnosis showed a WHO Grade 1 ‘raddled’ psammomatous meningioma with extensive metaplastic ossification. A literature review of 29 cases of intraparenchymal meningiomas regarding their clinical presentations, location and management was performed.
Meningiomas can be found in any region of the brain with and without dural attachment. Intraparenchymal meningiomas can have multiple entities mimicking their presentation. Caution must be used regarding the preoperative differential diagnosis.
PMCID: PMC4320455  PMID: 25547419
Meningiomas; Intraparenchymal meningiomas; Cavernous malformation; Ossification; ‘popcorn’; Hemosiderin deposits
14.  Superficial epithelioma with sebaceous differentiation involving the eyelid: a case report 
Superficial epithelioma with sebaceous differentiation is a rare benign epithelial neoplasm. It usually involves the head, neck or the back of a middle-age person. To the best of our knowledge, two ocular cases have been reported in the literature.
Case presentation
A 46-year-old man of Italian descent, with a known history of testicular seminoma treated by orchiectomy with chemotherapy and radiotherapy, presented with a tan-colored lesion measuring 4mm in diameter in his right upper lid that had been growing over 10 months. It was clinically diagnosed as papilloma. An excisional biopsy was done. On histological examination, the lesion was a well-circumscribed and sharply demarcated epithelial tumor attached to the overlying epidermis and characterized by plate-like proliferation of basaloid to squamous cells with clusters of mature sebaceous cells and foci of ductal differentiation. After a follow-up period of 5 months, no recurrence of the lesion has been documented.
Superficial epithelioma with sebaceous differentiation is part of the differential diagnoses of eyelid lesions. Arguments in the literature about the correct nomenclature of superficial epithelioma with sebaceous differentiation have resulted in under-diagnosed cases. The benign histological features and the lack of recurrence support its benign nature. Although no clear association has linked superficial epithelioma with sebaceous differentiation with Muir–Torre syndrome, further clinical correlation and close follow up for patients are recommended.
PMCID: PMC4320459  PMID: 25547332
Eyelid; Muir–Torre syndrome; Superficial epithelioma with sebaceous differentiation
15.  Head and neck myxofibrosarcoma: a case report and review of the literature 
Myxofibrosarcoma is the most common soft tissue sarcoma that occurs in late adult life, peaking in the seventh decade, and it is mainly encountered in the lower extremities. Myxofibrosarcoma of the head and neck are extremely rare. To the best of our knowledge, only 19 cases have been described in the head and neck so far. This is a literature review and retrospective chart review of our experience in head and neck myxofibrosarcoma treatment in our department.
Case presentation
In this case report we describe a 35-year-old Caucasian man who presented the first case of myxofibrosarcoma arising from the pterygopalatine fossa. The peculiar anatomical location and the extent in the midcheek region make this case a hard “challenge” for the surgeon, in order to guarantee wide surgical margins of resection. A total right maxillectomy was accomplished by means of the Weber-Ferguson approach, preserving the orbital floor. The excised portion was reconstructed using the free rectus abdominis myocutaneous flap. Postoperative radiotherapy was given to the area adjacent to the lesion, with a total dose of 60Gy. No relapse occurred in the 27-month postoperative follow-up.
The case described suggests the importance of combined surgical and adjuvant radiotherapy to avoid local and distant recurrences of the tumor. In our opinion, combined surgical and adjuvant radiotherapy followed by close clinical observation to search for a metastatic disease is advisable in all cases. Further studies are needed to confirm the efficacy of combined radio-chemotherapy for head and neck myxofibrosarcoma in terms of long-term disease-free survival.
PMCID: PMC4320505  PMID: 25547541
Malignant fibrous histiocytoma; Midcheek masses; Myxofibrosarcoma; Myxosarcoma
16.  Clinical and molecular findings in a Moroccan patient with popliteal pterygium syndrome: a case report 
Popliteal pterygium syndrome is a congenital malformation that includes orofacial, musculoskeletal and genitourinary anomalies. It is a rare autosomal dominant disorder due to a mutation of the IRF6 gene on 1q32.2.
Case presentation
A one-month-old Moroccan baby boy was diagnosed with typical features of popliteal pterygium syndrome and carried the c.250C>T; p.Arg84Cys mutation of the IRF6 gene.
We report on the first description of a Moroccan popliteal pterygium syndrome patient. This diagnosis allowed us to provide an appropriate course of management to the patient and offer genetic counseling to his family.
PMCID: PMC4320515  PMID: 25547932
Popliteal pterygium syndrome; Autosomal dominant; IRF6 gene
17.  Acquired haemophilia A associated with autoimmune thyroiditis: a case report 
Acquired haemophilia A is a rare life- and limb-threatening bleeding disorder if left untreated. Autoimmune thyroiditis is an autoimmune disorder that can be rarely associated with acquired haemophilia. Here we report a case of a 60-year-old woman presenting with cutaneous and muscle haematomas secondary to acquired haemophilia A in association with autoimmune thyroiditis, who was successfully treated with recombinant activated factor VII and immunosuppression.
Case presentation
A 60-year-old Sri Lankan woman with a background of longstanding hypothyroidism, diabetes mellitus, hypertension, hyperlipidaemia and bronchial asthma developed spontaneous cutaneous purpura and a limb-threatening intramuscular haematoma. Initial coagulation screening revealed prolonged activated partial thromboplastin time of 66.4 seconds (normal range 26 to -36 seconds) and time-dependent inhibitors against factor VIII. She had positive antinuclear antibody and antithyroid peroxidase (microsomal) antibody titre of over 1/80 and 1000IU/mL respectively. The diagnosis was therefore made of acquired haemophilia A in association with autoimmune thyroiditis. Acute limb-threatening bleeding was managed with recombinant activated factor VII (NovoSeven®). Immunosuppressive treatment consisting of oral prednisone 60mg/day and cyclophosphamide 100mg/day was administered in order to remove the factor VIII inhibitor. This treatment led to normalisation of her haemostatic parameters. This case illustrates a very rare association of acquired haemophilia and autoimmune thyroiditis as well as the importance of considering acquired haemophilia as a differential diagnosis of spontaneous bleeding.
Acquired haemophilia should be considered in the differential diagnosis of unexplained bleeding in adults. Treatment of the acute coagulopathy with recombinant activated factor VII and immunosuppressive therapy was successful in this case.
PMCID: PMC4320580  PMID: 25547669
Acquired haemophilia; Autoimmune thyroiditis; Bypassing agents; Factor VIII inhibitors; Haemophilia A; Immunosuppression
18.  Bowel obstruction caused by an internal hernia that developed after laparoscopic subtotal colectomy: a case report 
Laparoscopic surgery is a minimally invasive approach with good treatment outcomes and is currently the standard surgery for colorectal cancer in Japan. Mesenteric closure is considered unnecessary in laparoscopic colorectal surgery because it can damage the bowel and blood vessels. However, an internal hernia may develop if the mesentery is not repaired.
Case presentation
We report a case of internal hernia in a 61-year-old male of Japanese ethnicity. The patient had advanced sigmoid colon cancer, early-stage transverse colon cancer, and multiple adenomatous polyposis, and underwent laparoscopically-assisted subtotal colectomy. Bowel obstruction developed six days postoperatively and did not improve with conservative treatment. Abdominal computed tomography detected an internal hernia, prompting emergency surgery in which the ileum protruding into the mesenteric defect and an anastomotic stricture were detected. Reanastomosis, mesentery closure, and ileostomy were performed after hernia repair.
In this case, open surgery was necessary due to bowel obstruction after laparoscopic colectomy. This outcome indicated that mesenteric closure should have been performed. Thus, the benefits of mesenteric closure require assessment in future cases.
PMCID: PMC4320605  PMID: 25547813
Laparoscopic surgery; Subtotal colectomy; Bowel strangulation; Internal hernia; Mesenteric closure
19.  Granular cell tumor of the breast: a case report 
A granular cell tumor involving the breast parenchyma was first described by Abrikossoff in 1931. Localization of this lesion to the breast is very rare, accounting for between 5% and 15% of all granular cell tumor cases. We present this case because of the rarity of this tumor. It is frequently confused with breast carcinoma on clinical and radiological examination, and its diagnosis can therefore be challenging for clinicians, radiologists and pathologists.
Case presentation
We report the case of a 32-year-old Moroccan woman who presented with a palpable mass in her right breast. Mammography and ultrasound examination revealed a heterogeneous, irregular and poorly limited mass, located at the union of the outer quadrants of her right breast. The mass was in contact with her latissimus dorsi and suspicious for malignancy. A histological examination combined with immunohistochemical study revealed it to be a granular cell tumor.
Although a granular cell tumor of the breast is a rare breast neoplasm, it should be considered in the differential diagnosis of benign and malignant lesions. Pathologists should bear in mind a granular cell tumor when examining material containing cells with abundant granular cytoplasm to avoid misdiagnosing breast carcinoma, which could lead to unnecessary surgery.
PMCID: PMC4307888  PMID: 25541096
Breast; Granular cells tumor; Schwann cells; S-100 protein
20.  Respiratory tract isolation of Mycobacterium europaeum following influenza infection in an immunocompromised patient: a case report 
Mycobacterium europaeum, a slow-growing nontuberculous mycobacteria belonging to the Mycobacterium simiae complex, was described after the seminal characterization of five isolates collected from three sputum specimens and a jaw gland biopsy in Italy, Greece and Sweden. Five respiratory tract isolates were further reported in Iran. Here, we report the first isolation of M. europaeum in France, in the respiratory tract of a patient co-infected with human immunodeficiency virus and hepatitis C virus.
Case presentation
A 49-year-old Caucasian woman with a 26-year history of human immunodeficiency virus-hepatitis C virus co-infection was admitted for significant influenza-like syndrome in a context of repetitive exacerbations of chronic obstructive pulmonary disease. Significant biological parameters included lymphocytes of 1.6G/L including 237/mm3 T4 lymphocytes, a human immunodeficiency virus viral load of 1.6 log and a hepatitis C virus viral load of 6 log. Reverse-transcriptase polymerase chain reaction of her nasopharyngeal aspiration confirmed influenza A H1N1. Three sputum specimens lacked acid-fast bacilli but one grew mycobacteria identified by using matrix-assisted laser desorption ionization/time-of-flight mass spectrometry as M. europaeum with a 1.56 log score. A 1,482-bp 16S ribosomal ribonucleic acid gene sequence yielded 99% similarity with both Mycobacterium parascrofulaceum ATCC BAA-614 and M. europaeum DSM 45397T and partial rpoB polymerase chain reaction-sequencing yielded a 725-bp sequence exhibiting 100% similarity with M. europaeum strain DSM 45397T.
We report the first isolation of M. europaeum in France, in the respiratory tract of a patient co-infected with human immunodeficiency virus and hepatitis C virus. M. europaeum warrants further attention in immunosuppressed patients with influenza, using matrix-assisted laser desorption ionization/time-of-flight mass spectrometry and rpoB partial sequencing as tools for its accurate identification.
PMCID: PMC4307153  PMID: 25539638
HIV; Influenza; MALDI-TOF-MS; Mycobacterium europaeum; rpoB
21.  Polyorchidism: two case reports and a review of the literature 
Polyorchidism is a very rare anomaly that is defined by the presence of more than two testes. Although its presentation is primarily as triorchidism, cases of four testes have also been reported in the literature.
Case presentation
In this report, we describe color Doppler ultrasound findings in two cases. Patient 1 was a 37-year-old Arabic man with a scrotal mass and a double testicle in the right hemiscrotum visualized by ultrasound. Patient 2 was an 11-year-old Arabic boy with an inguinal mass resulted to be an additional testicle in the inguinal canal. The echogenic texture and vascular flow of supernumerary testicles in question were similar to those of the normal testicles; however, their size was smaller. After 3 years of follow-up, the tertiary testes in the two patients remained stable in both size and echogenicity.
Ultrasound plays a crucial role in the evaluation of masses. Notably, inguinal or scrotal masses should not always be considered as lymph nodes or tumors. Indeed, a radiologist should always keep polyorchidism in mind when such masses are encountered.
PMCID: PMC4307231  PMID: 25539715
Polyorchidism; Scrotum; Testes; Ultrasound
22.  Renal vein thrombosis in the post-partum period: a case report 
Physiological hypercoagulability is a known condition in pregnancy designed to limit the risk of bleeding; it may exceptionally be complicated by thrombosis of the renal vein. To the best of our knowledge, this is the third case of renal venous infarction reported in the literature.
Case presentation
We report the case of a 43-year-old Caucasian woman, a mother of three sons who presented with left flank pain and hematuria. The clinical investigations did not find any other cause for her thrombophilia.
Clinical onset is not specific, so it is important to evoke the diagnosis in the context of pregnancy; computed tomography angiography is the investigation of choice to set the diagnosis. It is important to know that anticoagulation therapy must be initiated as soon as possible.
PMCID: PMC4307671  PMID: 25539573
Flank pain; Hematuria; Hypercoagulability; Post-partum; Venous renal infarction
23.  Intrapartum anti-disseminated intravascular coagulation therapy leading to successful vaginal delivery following intrauterine fetal death caused by placental abruption: a case report 
Disseminated intravascular coagulation due to placental abruption with intrauterine fetal death is not uncommon. It can result in increased maternal mortality rates and the need for hysterectomy or greater transfusion volumes if the delivery is not completed within six to eight hours. However, consensus is lacking regarding the delivery approach for cases in which delivery is prolonged.
Case presentation
A 37-year-old Japanese woman was transported to our tertiary center two and a half hours after the onset of labor because of a diagnosis of placental abruption with intrauterine fetal death at 40 weeks and three days’ gestation. On arrival, although severe hypofibrinogenemia was observed, there was no external hemorrhage. Because her cervical canal dilation was good (Bishop score, 7), labor was induced using oxytocin. Anti-disseminated intravascular coagulation therapy was simultaneously started via transfusion. After her hypofibrinogenemia resolved, delivery progressed rapidly, and the fetus was delivered approximately 10 hours after the onset. To reduce postpartum hemorrhage, 6g of fibrinogen concentrate and tranexamic acid, an antifibrinolytic agent, were administered immediately before extraction of the dead fetus and placenta. Although the amount of intrapartum hemorrhage was 1824g, there was no abnormal bleeding after delivery, and our patient was discharged three days later.
In cases of placental abruption complicated with disseminated intravascular coagulation, intrapartum administration of coagulation factors can simultaneously promote effective labor and correct hypofibrinogenemia, enabling minimally invasive vaginal delivery.
PMCID: PMC4307188  PMID: 25532532
DIC; Intrauterine fetal death; Placental abruption; Vaginal delivery
24.  Anaplastic transformation of papillary thyroid carcinoma in multiple lung metastases presenting with a malignant pleural effusion: a case report 
Anaplastic transformation of well-differentiated papillary thyroid carcinoma at distant metastasis sites is rare. To the best of our knowledge, this is the first report of an autopsy case of anaplastic transformation of papillary thyroid carcinoma in multiple lung metastases presenting with a malignant pleural effusion.
Case presentation
We report an autopsy case of a 61-year-old Japanese man with anaplastic transformation of papillary thyroid carcinoma with multiple lung metastases presenting with a malignant pleural effusion, which was difficult to diagnose by cytological examination before the autopsy. He presented with a 1-month history of progressive dyspnea, and examination of the left pleural effusion revealed a bloody exudate with an increase in thyroglobulin; however, malignant cells in the pleural fluid were negative for thyroglobulin.
It is important to be aware that anaplastic transformation of differentiated thyroid carcinoma could develop in lung metastases and could be a cause of a malignant pleural effusion.
PMCID: PMC4307622  PMID: 25532447
Anaplastic transformation; Lung metastasis; Malignant pleural effusion; Papillary thyroid carcinoma
25.  Successful long-term management of choroidal neovascularization secondary to angioid streaks in a patient with pseudoxanthoma elasticum: a case report 
We describe the long-term effectiveness and tolerability of intravitreal vascular endothelial growth factor inhibitor ranibizumab in a patient with pseudoxanthoma elasticum with bilateral macular choroidal neovascularization secondary to angioid streaks.
Case presentation
A 54-year-old Caucasian man with history of heart disease presented with visual loss in his right eye. An examination revealed choroidal neovascularization and reduced visual acuity, while no abnormalities were seen in his left eye. He was diagnosed with angioid streaks associated with pseudoxanthoma elasticum. Off-label treatment with intravitreal bevacizumab once a month initiated in December 2007 was discontinued after 3 months due to lack of efficacy. In September 2008, the patient reported reduced visual acuity in his left eye and an examination revealed changes. Left eye treatment was initiated in October 2008 with a loading dose (three consecutive monthly intravitreal injections of ranibizumab 0.5mg/50μL) followed by 0.5mg/50μL followed by treatment as needed until May 2014. After 21 ranibizumab injections, an examination revealed angioid streaks and choroidal neovascularization in both eyes. His right eye showed retinal layer deterioration with outer limiting membrane and photoreceptor inner/outer segment junction involvement. His left eye had a smaller foveal scar, with other areas preserved. Visual acuity was stable in his treated left eye, but had deteriorated in his right eye. Ranibizumab treatment was well tolerated with no adverse events reported.
In the present case, an as-needed regimen of ranibizumab after an initial loading dose, achieved maintenance of visual function and was well tolerated over a period of almost 6 years in a patient with pseudoxanthoma elasticum and high cardiovascular risk. As anti-vascular endothelial growth factor agents are associated with increased risk of systemic effects, particularly arterial thromboembolic events, following intravenous administration, the absence of serious thromboembolic or cardiovascular adverse events throughout the 6-year treatment period is particularly encouraging considering our patient’s high cardiovascular risk status.
PMCID: PMC4308002  PMID: 25529762
Angioid streaks; Bevacizumab; Choroidal neovascularization; Pseudoxanthoma elasticum; Ranibizumab

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