Problems of live virus vaccine-associated poliomyelitis a paralytic case with isolation of all three poliovirus types

The case of a 4-month-old girl is described who developed a paralytic polio-like syndrome 3 weeks after oral polio vaccination (OPV). All three poliovirus types could be isolated (9 days after onset of disease polio type 2, and 33 days after onset of disease types 1 and 3, respectively).In order to classify these isolates as Sabin (vaccine)-like (SL) or non-Sabin-like (non-SL), several markers were tested in three laboratories [intratypic serodifferentiation, reproductive capacity at supraoptimal temperature (RTC), A1(OH)₃ gel elution assay, and oligonucleotide mapping]. The results of the marker determinations were not uniform, but — summarizing all data — it seems plausible to associate the disease with the OPV. The significance of marker determinations in proving a vaccine-induced poliomyelitis is discussed in the light of this clinical case. Some comments are made on poliovirus vaccination policy in developed countries.     

Three cases of paralytic poliomyelitis associated with type 3 vaccine poliovirus strains in Bulgaria

Oral poliovirus vaccine (OPV) can cause, in extremely rare cases vaccine‐associated paralytic poliomyelitis in recipients, or contacts of vaccinees. Three cases of vaccine‐associated paralytic poliomyelitis (two contacts and one recipient) occurred in the Bourgas region of Bulgaria in the spring of 2006. The first two cases, notified as acute flaccid paralysis, were 55 days old unvaccinated twin brothers, having been in contact with vaccinees. The third case concerned a 4‐month‐old infant who had received the first OPV dose 37 days prior to the onset of illness. Complete clinical, epidemiological, virological, serological and molecular investigations of the children with paralysis and their contacts were undertaken. In all the three cases type 3 polioviruses were isolated from fecal samples and characterized as Sabin‐like poliovirus strains. Type 3 polioviruses isolated from the twin brothers demonstrated by sequence analysis U‐to‐C back mutation at nt 472 of the 5′ UTR, known to correlate with neurovirulence, and mutation in the VP1 region. Type 3 poliovirus isolated from the third child demonstrated in the 3D sequenced region a recombination with Sabin type 1 poliovirus. In the latter region, three silent mutations and one, resulting in amino acid substitution, were also observed. The clinical, epidemiological and virological data and the neurological sequelae observed 60 days following the onset of paralysis, confirmed the diagnosis of vaccine‐associated paralytic poliomyelitis in all the three patients. J. Med. Virol. 81:1661–1667, 2009. © 2009 Wiley‐Liss, Inc.     

Combined hepatocellular-cholangiocarcinoma after tetralogy of Fallot repair: a case report and review of literature

BackgroundLiver fibrosis and cancer are serious hepatic complications for patients with congenital heart diseases. We present a rare case of combined hepatocellular carcinoma (HCC) and cholangiocarcinoma (CCA) (cHCC-CCA) after the repair of tetralogy of Fallot (TOF).Case presentationA 54-year-old Japanese woman had undergone biventricular repair for TOF at 7 years old. She presented with abdominal distension. Abdominal CT revealed ascites and a 90-mm tumor involving the liver's left lobe. Tumor marker values were: alpha-fetoprotein, 16,208 ng/mL and des-gamma-carboxy prothrombin, 33,920 mAU/mL. The preoperative diagnosis was malignant tumor of the liver (e.g., HCC or intrahepatic CCA). We performed a left lobectomy of the liver. Histopathologically, the tumor was composed of two components growing in trabecular and irregular tubular patterns accompanied by a transitional area; the tumor was diagnosed as cHCC-CCA. The non-cancerous area showed fibrous change mainly surrounding a central vein and sinusoid, expanding toward the portal area without inflammation.ConclusionsWe provide the details of our patient's cHCC-CCA that developed from fibrous congestive liver associated with right-sided heart failure after TOF repair, diagnosed based on histopathological features. We discuss liver fibrosis as a hepatic complication and a careful follow-up maneuver for improving the outcomes of patients with chronic hepatic congestion.     

Lateral ventricular meningioma presenting with intraventricular hemorrhage: a case report and literature review

Lateral ventricular meningiomas presenting with primary intraventricular hemorrhage are extremely uncommon. We report here a case of primary intraventricular hemorrhage attributable to a lateral ventricular meningioma. This case concerns a 46-year-old female patient who presented with sudden onset of headache. Computed tomography (CT), computed tomography angiography (CTA) and magnetic resonance imaging (MRI) examinations showed hemorrhage from a ruptured tumor mass, which was pathologically confirmed as a transitional meningioma. The patient underwent surgical treatment and had a good prognosis. A retrospective review of eight previous cases of hemorrhage from ruptured lateral ventricular meningiomas revealed that hemorrhage of lateral ventricular meningiomas and hemorrhage of meningiomas at other intracranial sites have similar causes. The clinical and pathological features of ruptured lateral ventricular meningiomas are consistent with those of unruptured lateral ventricular meningiomas. As this clinical entity is extremely rare, attention is called for while performing differential diagnosis.     

Extraosseous osteosarcoma presenting with intestinal hemorrhage: case report and literature review

A 23-year-old man presented with intestinal bleeding due to an extraosseous osteosarcoma of the jejunum. A lesion was also found in the deltoid muscle, and other metachronous soft tissue sites developed subsequently. The presence of malignant osteoid was documented by immunohistochemical studies of one of the lesions. The patient died of metastatic disease 19 months after diagnosis, despite surgical resections and adjuvant chemotherapy. This unique presentation is discussed, and the literature concerning extraosseous osteosarcoma is reviewed.     

Prostate cancer presenting as an endobronchial mass: a case report with literature review

Endobronchial metastasis from a prostate cancer is uncommon. Diagnosis of prostate carcinoma after primary presentation with an endobronchial mass is very rare. The authors describe the case of an 84-year-old man with endobronchial metastases from prostatic carcinoma presenting primarily with pulmonary symptoms. The diagnosis of prostate cancer and endobronchial metastases was made by a bronchoscopic bronchial biopsy and confirmed by immunohistological staining with prostate-specific antigen. The importance of this manifestation along with clinical and therapeutic implications is discussed here.     

Secondary intestinal amyloidosis presenting intractable hematochezia: a case report and literature review

Amyloidosis is characterized by an extracellular deposition of insoluble fibrils. Amyloid deposition caused various clinical symptoms associated with affected organs. Secondary amyloidosis without renal involvement and chronic inflammatory conditions is rarely reported. We experienced a case of secondary intestinal amyloidosis presented with recurrent hematochezia and abdominal pain in a 54-year-old male. Sigmoidoscopy and abdominal computed tomography (CT) presented ischemic colitis and necrosis of whole colon. On microscopically, pinkish amorphous materials were infiltrated in the lamina propria and the thickened submucosal vessel walls in colon. The apple-green birefringence with polarized light on Congo red stain was demonstrated in the lamina propria and submucosal vessel walls. The deposits were positive for amyloid A and κ and negative for λ. The echocardiography and cardiac MRI findings showed infiltratives cardiomyopathy involving amyloidosis. Despite of conservative treatment, ischemic colitis and hemorrhage were aggravated and the patient expired.     

Vaccine-associated paralytic poliomyelitis: a case report of flaccid monoparesis after oral polio vaccine

This report describes a case of acute flaccid paralysis after administration of oral polio vaccine (OPV). A 4 month-old male patient with the decreased movement of left lower extremity for 1 month was transferred to the Department of Pediatrics. He received OPV with DTaP at 2 months of age. Flaccid paralysis was detected 4 weeks after OPV immunization. Attempts to isolate Sabin-like viruses in the two stool and CSF samples failed because those specimens were collected more than 2 month after the onset of paralysis. Hypotonic monoparesis (GIV/V), hypotonia and atrophy on the left lower extremity, and ipsilateral claw foot persisted for more than 18 months, while we followed him with rehabilitation therapy. This is the first case of officially approved, recipient vaccine-associated paralytic poliomyelitis in Korea.     

Neurologically presenting Whipple disease: case report and review of the literature

A previously healthy male with subacute onset right leg weakness was suspected to have an astrocytoma as imaging showed a lesion. Subsequent biopsy showed the presence of foamy macrophages containing periodic acid-Schiff staining granules, suggesting Whipple disease as a possible diagnosis.     

Gastric adenocarcinoma deposits presenting as multiple cutaneous nodules: a case report with review of literature

Metastases to the skin from internal tumors are uncommon, yet they may be the first presentation of such malignancies. They usually arise from the breast, lung and large bowel. Cutaneous metastases from gastric adenocarcinoma are extremely rare. We report a case of gastric adenocarcinoma that at presentation had multiple clinically benign looking skin nodules. Fine needle aspiration cytology of the cutaneous nodule revealed metastatic deposits. The current literature on cutaneous metastases of gastric carcinoma is reviewed in this case report.     

Metastatic adenocarcinoma presenting as an inguinal hernia: a case report and review of literature

A 61 year old man presented with an inguinal hernia with no other significant symptoms. Histopathological examination of the hernial sac revealed metastatic deposits of a mucin secreting adenocarcinoma which was confirmed by subsequent tumor marker levels. Patient was put on chemotherapy for disseminated adenocarcinoma and is tolerating it well. This case emphasizes the need to carefully examine all hernial sacs received for pathological examination.     

Giant intrasellar aneurysm presenting with panhypopituitarism and subarachnoid hemorrhage: case report and literature review

A 52-year-old woman was admitted to our hospital presenting with subarachnoid hemorrhage, left ophthalmoplegia, and right hemiparesis. Previous symptoms and signs suggested the presence of panhypopituitarism. A giant intracranial aneurysm of the internal carotid artery, diagnosed by magnetic resonance imaging, showed the characteristic flow void phenomenon with black appearance. Analysis of endocrine function disclosed panhypopituitarism and hyperprolactinemia. After proximal balloon occlusion of the aneurysm, diabetes insipidus developed. Pituitary function reassessed 6 months after initial admission confirmed anterior and posterior hypopituitarism and hyperprolactinemia. Possible mechanisms are discussed. A review of the literature on pituitary dysfunction caused by carotid artery aneurysms discloses that the pituitary-gonadal axis is the most frequently involved (67.5% of cases), followed by the pituitary-adrenal axis (48.6%) and the pituitary-thyroid axis (40.5%). These frequencies are very similar to those described in other types of hypopituitarism.Abbreviations ICA internal carotid artery - MRI magnetic resonance imaging - SAH subarachnoid hemorrhage Correspondence to: J.M. Fernandez-Real     

Cardiac aspergillosis in patients with acquired immunodeficiency syndrome: a case report and review of the literature

Cardiac aspergillosis is uncommon in patients with acquired immunodeficiency syndrome (AIDS) in the absence of open heart surgery. We report a unique case of a 62-year-old man with AIDS who developed Aspergillus pancarditis with Aspergillus vegetations on mitral valve without evidence of pulmonary aspergillosis. There was extensive embolization to the brain and multiple foci of Aspergillus infection in kidneys and adrenal glands. There are only 10 documented cases of cardiac aspergillosis in the literature (1966-2003) in severely immunocompromised AIDS patients with CD4 T-lymphocyte counts ranging from 10 to 121 cells/muL. The cardiac aspergillosis could result from invasive pulmonary aspergillosis, either by hematogenous dissemination or by direct invasion, and skin Aspergillus infection can be carried through the bloodstream to the right heart in intravenous drug abusers. Most of the reported cases of cardiac aspergillosis were diagnosed at autopsy. Mortality among AIDS patients with cardiac aspergillosis is 100%, despite appropriate therapy.     

Primary retroperitoneal dysgerminoma presenting as an adrenal tumor: a case report and literature review

Primary extragonadal germ cell tumors are rare and mostly occur in young men with predominance of nonseminomatous histology. We report an undescribed case of primary retroperitoneal dysgerminoma presenting as an adrenal tumor in a 17-year-old girl. Surgery was performed on a 10 × 9.5 cm sized adrenal gland tumor and the resected tumor showed unequivocal histological features of dysgerminoma. The diagnosis was confirmed by the tumor's germ cell immunophenotype. Postoperative ultrasonography, CT and PET over a 6-month period revealed no evidence of ovarian lesion. The patient is stable, but with a suspicious residual tumor after adjuvant chemotherapy.     

Proportions of immunoglobulin isotypes in paralytic poliomyelitis and after vaccination

Immunoglobulin isotype composition of poliovirus antibodies was studied by isotype-specific solid-phase radio-immunoassay (RIA) in four patients with paralytic poliomyelitis, five adults receiving live poliovirus vaccine as a booster immunization, and seven children receiving first doses of inactivated poliovirus vaccine. In paralytic poliomyelitis serum and cerebrospinal flind (CSF) poliovirus antibodies were mainly of IgG1, IgG3, and IgA isotypes. IgM antibodies were found in sera but not in CSF. Either IgG2 and IgG4 antibodies were undetectable or the titers were low. In adults who had received live trivalent poliovirus vaccine, antibodies against poliovirus type 3 were detected in IgG1 (53% of total antibodies), IgG3 (25%), IgM (9%), IgA (8%), IgG2 (3%), and IgG4 (2%) isotypes. In prevaccination and late postvaccination sera the share of IgG3 antibodies was exceptionally high (35%). In children who received inactivated poliovirus vaccine, antibodies developed in IgG1 (53–61% of total antibodies for poliovirus types 1, 2, and 3), IgG3 (12–21%), and IgM (23–33%) isotypes. Antibody levels in IgG2, IgG4, and IgA isotypes were low and observed only in a few cases. Like other viral antibodies IgG1 and IgG3 isotypes were the major IgG subclasses in poliovirus antibodies.     

Intravascular large B-cell lymphoma presenting as cholecystitis and pancytopenia: case report with literature review

Intravascular large B-cell lymphoma (IVLBCL) is a rare variant of extranodal diffuse large Bcell lymphoma with only a few more than 300 cases reported. It is characterized as lymphoma cells confined to the lumina of small vessels, so patients usually do not present with masses or lymphadenopathy. Clinical presentations of these patients are non-specific and the pathologic changes may be subtle, which often leads to delayed diagnoses and, in many instances, a postmortem diagnosis. IVLBCL can essentially involve the vessels of any organ, but it is quite rare for the gallbladder to serve as the initial presenting site; there are only four such cases reported in the English literature. Furthermore, IVLBCL of the gallbladder with peripheral blood involvement is even less common. We report a recent case of IVLBCL presenting as acute cholecystitis and pancytopenia. The patient underwent a simple cholecystectomy. Examination of the gallbladder showed clusters of large lymphoma cells within lumina of small vessels in the gallbladder wall. These cells were positive for CD5/CD20 and negative for CD3, CD10, and TdT. Based on these findings, a diagnosis of IVLBCL was made. Coincidently, circulating lymphoma cells were identified in the peripheral blood and confirmed by flow cytometric analysis (positive for CD19/CD20/CD5, without light chain expression). The patient was started on chemotherapy but subsequently died of chemotherapy related multi-organ failure 10 days after the initial diagnosis.     

Ala67Thr mutation in the poliovirus receptor CD155 is a potential risk factor for vaccine and wild‐type paralytic poliomyelitis

Poliovirus infections can be asymptomatic or cause severe paralysis. Why some individuals develop paralytic poliomyelitis is unknown, but a role for host genetic factors has been suggested. To investigate if a polymorphism, Ala67Thr, in the poliovirus receptor, which has been found to facilitate increased resistance against poliovirus‐induced cell lysis and apoptosis, is associated with increased risk of paralytic poliomyelitis, poliovirus receptor genotyping was undertaken among Italian subjects with vaccine‐associated (n = 9), or with wild‐type paralytic poliomyelitis (n = 6), and control subjects (n = 71), using RFLP‐PCR and pyrosequencing. Heterozygous poliovirus receptor Ala67Thr genotype was found in 13.3% of the patients with paresis and in 8.5% of the controls (Odds Ratio = 1.667). The frequency of Ala67Thr among the controls is in agreement with earlier published data. It is concluded that the Ala67Thr mutation in the poliovirus receptor is a possible risk factor for the development of vaccine‐associated or paralytic poliomyelitis associated with wild‐type virus. J. Med. Virol. 81:933–936, 2009. © 2009 Wiley‐Liss, Inc.     

CTLA-4 gene mutation and multiple sclerosis: A case report and literature review

We reported a patient with autoimmunity (multiple sclerosis), immunodeficiency (hypogammaglobulinemia with severe infections), enteropathy (diarrhea with intestinal inflammation), splenomegaly, lymphadenopathy and lymphocytic infiltration of non-lymphoid organs (lung, gut and brain). The patient was found to have a heterozygous mutation in cytotoxic T lymphocyte antigen-4, and had excellent response to abatacept.     

Painless angioleiomyoma: a case report with review of literature

A 55 year old male, who presented with a painless swelling of six months duration on his thigh, was clinically diagnosed to have a benign soft tissue neoplasm. Histological examination of the excision biopsy specimen revealed a benign tumour of the vascular smooth muscle, which was diagnosed as angioleiomyoma. Angioleiomyomas are painful lesions that can occur anywhere in the body. We present this case for its atypical clinical presentation and discuss its histologic features with a review of literature.