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case 203: gorham disease.a 77-year-old woman presented with pain in the right elbow of several years duration. there was no history of trauma, immunocompromise, or known malignancy. blood chemistry tests revealed an alkaline phosphatase level of 93 u/l (1.6 μkat/l) (30-120 u/l [0.5-2.0 μkat/l]), a sodium level of 139 mmol/l (135-147 mmol/l), a potassium level of 4.4 mmol/l (3.4-5.0 mmol/l), a calcium level of 2.38 mmol/l (2.2-2.55 mmol/l), a phosphate level of 1.81 mmol/l (0.85-1.5 mmol/l), a c-reactive protein level of ...201424568708
lectotype designations and nomenclatural changes in xylographus mellié (coleoptera, ciidae).we designate lectotypes and propose nomenclatural changes in xylographus mellié (coleoptera, ciidae) based on type specimens deposited in the museum of comparative zoology (usa), museum für naturkunde berlin (germany), the natural history museum (uk), muséum d'histoire naturelle de la ville de genève (switzerland), muséum national d'histoire naturelle (france), naturhistoriska riksmuseet (sweden) and naturhistorisches museum wien (austria). we designate lectotypes for the following species: cis ...201424493963
the phenolic content and its involvement in the graft incompatibility process of various pear rootstocks (pyrus communis l.).this study investigates the influence of various rootstocks for pear on the phytochemical composition in the phloem above and below the graft union and the role of phenols in pear graft incompatibility. assays of phloem with cambium from 4-year-old 'conference', 'abate fetel' and 'williams' pear trees grafted on different rootstocks: quince ma, quince ba 29, fox 11, farold 40 (daygon), seedling pyrus communis l. and own rooted (p. communis l.) were analyzed with hplc-ms. the most abundant phenol ...201424484960
localization of polysaccharides in isolated and intact cuticles of eucalypt, poplar and pear leaves by enzyme-gold labelling.the presence and characteristics of cuticle polysaccharides have been demonstrated by staining and spectroscopic methods, but their location in the cuticle remains unclear. furthermore, according to the prevailing model, polysaccharides are believed to be restricted to the cuticular layer and absent in the cuticle proper. with the aim of gaining insight into cuticular ultra-structure focussing on polysaccharides, cellulose and pectins have been identified and located in the transversal sections ...201424445334
effect of different packaging materials and storage intervals on physical and biochemical characteristics of pear.the fruits of semi-soft pear (pyrus communis) cv. punjab beauty harvested at physiologically mature stage in 3rd week of july were packed in corrugated fiberboard boxes (cfb) with low density polyethylene (ldpe) liners, cfb with high density polyethylene (hdpe) liners, crates with ldpe liners, crates with hdpe liners, cfb, crates and wooden boxes and stored in walk-in-cool chamber at 0-1 °c and 90-95% rh. the fruits were analyzed after 30, 45, 60 and 75 days for physiological loss in weight (plw ...201324425900
diffuse lymphangiomatosis: gorham-stout syndrome. 201424390534
the relationship between the morphology and structure and the quality of fruits of two pear cultivars (pyrus communis l.) during their development and maturation.the flavour and nutritional values of pears are appreciated by consumers worldwide, who, however, demand specific fruit quality, that is, attractive appearance, firmness and flavour, and health safety as well as long-term shelf life and storability. pear cultivars differ in terms of the above-mentioned traits; therefore, we undertook investigations to demonstrate the differences in structure of fruits of two pear cultivars that determine fruit quality in its broadest sense. the micromorphology, ...201324327806
a case of an idiopathic massive osteolysis with skip lesions.a patient with a 2-year history of pain in the left arm, and decreased strengths unrelieved by non-steroidal anti-inflammatory therapy, was being referred for repeating radiography. the radiologic examinations have demonstrated a unique pattern of non-contiguous osteolysis in the left elbow, proximal and distal radius, ulna, wrist, carpal bones, proximal and distal metacarpals and phalanges. multi-site biopsies were being performed and confirmed the diagnosis of massive osteolysis. to our knowle ...201624265571
combined effect of temperature and controlled atmosphere on storage and shelf-life of 'rocha' pear treated with 1-methylcyclopropene.the combination of temperature and atmosphere composition for storage of pyrus communis l. 'rocha' treated with 1-methylcyclopropene was investigated. fruits treated with 312 nl l(-1) 1-methylcyclopropene were stored at 0 ℃ and 2.5 ℃ in air and controlled atmosphere (ca) (3.04 kpa o2+ 0.91 kpa co2). fruits were removed from storage after 14, 26 and 35 weeks, transferred to shelf-life at approximately 22 ℃ and assessed for ripening and quality, symptoms of superficial scald and internal browning ...201524216324
novel molecular pathways in gorham disease: implications for treatment.rapid advances in evidence-based treatment schedules are a hallmark of modern oncology. in rare neoplastic diseases, however, clinical expertise is hard to build and evidence based on randomized trials almost impossible to collect. gorham disease is a rare form of lymphatic proliferation accompanied by osteolysis, which usually occurs in young adults. despite the fact that the clinical course of gorham disease is often devastating and occasionally fatal, insights into its biological background a ...201424214028
gorham disease of the lumbar spine with an abdominal aortic aneurysm: a case report.reports of gorham disease of the lumbar spine complicated by abdominal aortic aneurysms are rare.201424161363
taxonomic and nomenclatural notes on the genera themus motschulsky and lycocerus gorham (coleoptera, cantharidae).themus (s.str.) regalis (gorham, 1889), nom. rest.; themus (s.str.) scutulatus wittmer, 1983 = themus (s.str.) hmong kazantsev, 2007, syn. n.; themus (telephorops) coelestis (gorham, 1889) = themus violetipennis wang & yang, 1992, syn. n.; themus (telephorops) uniformis wittmer, 1983, stat. n. = themus (telephorops) cribripennis wittmer, 1983, syn. n.; themus (haplothemus) licenti pic, 1938, stat. rev., resurrected from synonymy with themus coriaceipennis (fairmaire, 1889); lycocerus aenescens ( ...201324146589
gorham's disease of spine.gorham's disease is a rare disorder characterized by clinical and radiological disappearance of bone by proliferation of non-neoplastic vascular tissue. the disease was first reported by jackson in 1838 in a boneless arm. the disease was then described in detail in 1955 by gorham and stout. since then, about 200 cases have been reported in the literature, with only about 28 cases involving the spine. we report 2 cases of gorham's disease involving the spine and review related literature to gain ...201324066223
propranolol as a treatment option in gorham-stout syndrome: a case report.gorham-stout syndrome is a very rare syndrome characterized by progressive angiomatosis and lymphangiomas involving multiple organs. we describe herein a girl with progressive gorham-stout syndrome since the age of 13 years. her disease involved the mediastinum, pericardium, vertebrae, ribs, and skull and did not respond to interferon and bisphosphonates. propranolol administration was initiated at the age of 18 years and was associated with improvement in pulmonary function tests and involution ...201424000203
global changes in dna methylation in seeds and seedlings of pyrus communis after seed desiccation and storage.the effects of storage and deep desiccation on structural changes of dna in orthodox seeds are poorly characterized. in this study we analyzed the 5-methylcytosine (m(5)c) global content of dna isolated from seeds of common pear (pyrus communis l.) that had been subjected to extreme desiccation, and the seedlings derived from these seeds. germination and seedling emergence tests were applied to determine seed viability after their desiccation. in parallel, analysis of the global content of m(5)c ...201323940629
vanishing bone disease of the mandible - a case report.massive osteolysis (gorham-stout syndrome) is a rare condition of unknown etiology that is thought to result from a localized endothelial proliferation of lymphatic vessels resulting in destruction and absorption of bone. osteolysis of the jaws has been reported in association with infection, cysts, neoplasia, and metabolic, endocrine, or hematologic abnormalities. rare cases of idiopathic osteolysis have also been recorded. a detailed review from various peer reviewed journals has been discusse ...201323924511
gorham-stout syndrome in mainland china: a case series of 67 patients and review of the literature.gorham-stout syndrome (gss) is a rare disorder of uncertain etiology and unpredictable prognosis. this study aims to present a comprehensive understanding of this rare entity.201323897792
gorham-stout syndrome affecting the temporal bone with cerebrospinal fluid leakage.gorham-stout syndrome is a rare disorder characterized by progressive osteolysis that leads to the disappearance of bone. lymphvascular proliferation causes the local destruction of bony tissue. owing to the low incidence of this syndrome, little is known about its etiology or treatment. we present an 11-year-old girl with gorham-stout syndrome that involved right petrous apex in temporal bone and upper clivus, which cause intracranial pressure increase and cerebrospinal fluid (csf) leakage. the ...201323827687
[enigmatic lymphatic diseases involving the lung].lymphedema associated with other developmental malformations (milroy syndrome, hennekam syndrome, noonan syndrome, gorham-stout syndrome, yellow nail syndrome) are unfrequent disease, but explorations led to the identification of genetic mutations that have then been validated in mouse models. however, lymphatic vessels complexity and its proximity with the venous system suggest the need for further researches, especially in the comprehension of pulmonary symptoms.201323561737
gorham syndrome with postoperative respiratory failure and requiring prolonged mechanical ventilation.gorham syndrome is a rare disease that presents as progressive osteolysis, and may affect any part of the skeleton. the pathologic process involves the replacement of normal bone by aggressively expanding but non-neoplastic vascular tissue, resulting in massive osteolysis of the adjacent bone. if the spine and ribs are affected, the subsequent kyphosis and chest wall deformity may cause severe restrictive ventilatory impairment. we report a 34-year-old male with gorham syndrome presenting as pro ...201323550170
gorham disease of the craniocervical junction: x-ray, computed tomography, and magnetic resonance imaging findings.gorham disease of massive osteolysis is a spontaneous, idiopathic, and progressive form of primary osteolysis. it has no age, sex, or race predilection, and patients are mostly asymptomatic until severe deformity or pathological fracture becomes evident.201323462515
gorham disease of the proximal femur: a case report and review of published reports. 201323420753
distribution of transglutaminase in pear pollen tubes in relation to cytoskeleton and membrane dynamics.transglutaminases (tgases) are ubiquitous enzymes that take part in a variety of cellular functions. in the pollen tube, cytoplasmic tgases are likely to be involved in the incorporation of primary amines at selected peptide-bound glutamine residues of cytosolic proteins (including actin and tubulin), while cell wall-associated tgases are believed to regulate pollen tube growth. using immunological probes, we identified tgases associated with different subcellular compartments (cytosol, membrane ...201323396835
unspecific, idiopathic isolated osteolysis (gorham-stout syndrome) of the mandibular condylar process with its radiological, histological and clinical features: a case report and review of literature.the unspecific, idiopathic isolated osteolysis is a rare condition in the various known osteopathies and better known as gorham-stout syndrome (gorham's disease, gd). it belongs to the primary idiopathic osteolysis, and its aetiology is poorly understood. many different localisations have been described, but there are only 48 cases affecting the maxillofacial region until now.201423378037
gorham-stout disease and generalized lymphatic anomaly--clinical, radiologic, and histologic differentiation.gorham-stout disease (gsd) is a rare vascular disorder of lymphatic origin characterized by progressive osteolysis. generalized lymphatic anomaly (gla) is a multisystem disorder that also commonly affects bone. we hypothesized that gorham-stout disease is different from other osseous lymphatic anomalies. we proposed to discriminate these entities by analyzing findings on skeletal imaging.201323371338
syndromes associated with vascular tumors and malformations: a pictorial review.use of the international society for the study of vascular anomalies (issva) classification system has been strongly recommended in recent years because of the need for separate therapeutic measures for patients with vascular tumors and malformations. in the issva classification system, vascular tumors, which are neoplastic, are distinguished from vascular malformations, which are caused by vascular structural anomalies and are not neoplastic, on the basis of the presence or absence of neoplasti ...201323322836
gorham-stout syndrome affecting the left mandible: a case report.gorham-stout syndrome is an extremely rare condition in which spontaneous, progressive resorption of bone occurs. owing to its low incidence and variable clinical presentation, the diagnosis is often missed or delayed, and at present, there are no specific guidelines for its treatment. we present the case of a 20-year-old male diagnosed with gorham-stout syndrome with involvement of the left mandible, and discuss its diagnostic and therapeutic features.201323251259
spotlight on the safety net. a community collaboration. lucille w gorham intergenerational community center (igcc). 201223189440
gorham-stout syndrome presenting in a 5-year-old girl with a successful bisphosphonate therapeutic effect.gorham-stout syndrome (gss), also known as gorham-stout disease, massive osteolysis, disappearing bone disease or phantom bone, is a rare disorder of the musculo-skeletal system. it most commonly involves the skull, shoulder and pelvic girdle. histological examination reveals a progressive osteolysis always associated with an angiomatosis of blood vessels and sometimes of lymphatics, which seemingly is responsible for the destruction of the bone. it is extremely rare that gorham-stout syndrome i ...201223181116
gorham-stout syndrome of the facial bones: a review of pathogenesis and treatment modalities and report of a case with a rare cutaneous manifestations.gorham disease is a very rare condition associated with spontaneous destruction and resorption of 1 or more bones anywhere in the body. many authors have suggested and/or implicated trauma as the initiating factor in the majority of the reported cases. it can affect almost all bones, and a combination of bones has been reported. in the maxillofacial skeleton, the first facial case was reported by romer in 1928. until now, only a few cases of gorham disease affecting the maxillofacial bones, incl ...201223159123
[gorham-stout syndrome--a pediatric clinical case]. 201223149645
development of cultivar-specific dna markers based on retrotransposon-based insertional polymorphism in japanese pear.we developed retrotransposon-based insertional polymorphism (rbip) markers based on the long terminal repeat (ltr) sequences of copia-like retrotransposon ppcrt4 and flanking genome sequences, which were derived from 454 sequencing data from japanese pear (pyrus pyrifolia) 'hosui'. out of 40 sequences including both ltr and flanking genome regions, we developed 22 rbip markers and used them for dna profiling of 80 pear cultivars: 64 japanese, 10 chinese (pyrus ussuriensis) and 6 european (pyrus ...201223136514
differential expression of anthocyanin biosynthetic genes and transcription factor pcmyb10 in pears (pyrus communis l.).anthocyanin biosynthesis in various plants is affected by environmental conditions and controlled by the transcription level of the corresponding genes. in pears (pyrus communis cv. 'wujiuxiang'), anthocyanin biosynthesis is significantly induced during low temperature storage compared with that at room temperature. we further examined the transcriptional levels of anthocyanin biosynthetic genes in 'wujiuxiang' pears during developmental ripening and temperature-induced storage. the expression o ...201223029391
gorham disease of mandible treated with post-operative radiotherapy. 201224455523
constituents of twig bark of pear cultivars (pyrus species).organic solvent extracts from fresh twig bark of japanese pear cultivars (pyrus serotina) shinko and nijisseiki, and european pear cultivar (p. communis) le lectier were obtained by maceration with n-hexane and etoac, and analyzed in gc-eims experiments. in these two japanese cultivars, the lupeol, betulin, epifriedelinol, friedelin and arbutin contents of nijisseiki were higher than those of shinko. in the case of the lupane-type triterpenes, lupeol and betulin, the lupeol content of japanese p ...201222978211
gorham-stout disease as a complication of posterior shoulder capsulorrhaphy. 201222889527
successful management of intractable chylothorax in gorham-stout disease by awake thoracoscopic surgery.gorham-stout is an extremely rare disease, which is characterized by proliferation of vascular and lymphatic bone structures. a 15-year-old male patient was the diagnosis of gorham-stout disease of the cervical spine with chylothorax. awake thoracoscopic ablation was performed using bronchoscopic tools and awake thoracoscopic debridement of the thoracoscopic cavities and chemical pleurodesis with ok-432 were repeated. the amount of drained liquid was controlled. there was no recurrence of pleura ...201322865280
invasive lymphatic malformation (gorham-stout) of the pelvis with prominent skin involvement.gorham-stout syndrome is a rare disease characterized by progressive osteolysis leading to disappearance of the bone. vascular proliferations have been implicated in the pathogenesis of this syndrome. the case of a 7-year-old girl with a prominent invasive lymphatic malformation on the lumbosacral area and massive osteolysis of the pelvic girdle is reported.201622823281
gorham disease in the maxilla.gorham disease is a rare condition that is characterized by the proliferation of thin-walled vascular channels associated with regional osteolysis. the exact etiology of gorham disease is unknown. the diagnosis of gorham disease is based on clinical, radiological, and histological features after excluding osteolysis, which is secondary to other pathologic processes. those pathologic processes include congenital, metabolic, neoplastic, and immunologic etiologies and infections. the appearance of ...201222801155
pten hamartoma tumor syndrome and gorham-stout phenomenon.pten: hamartoma tumor syndrome (phts) is a group of syndromes caused by mutations in pten. gorham-stout phenomenon (gsp) is a rare condition characterized by proliferation of vascular structures in bones, resulting in progressive osteolysis. here we present a 1-year-old boy with phts and gsp. the lesion that later proved to be gsp was evident from the age of 4 months, and became symptomatic at the age of 1 year. eventually, he developed a fatal chylothorax. mutation analysis revealed a germline ...201222628360
[anaesthetic implications of gorham syndrome]. 201222480560
gorham-stout disease and cerebrospinal fluid otorrhea.gorham-stout disease is a rare entity characterized by vascular proliferation causing local destruction of bone tissue. owing to its low incidence and variable clinical presentation, the diagnosis requires a high degree of awareness by the clinician.201122456030
treatment of early-onset gorham syndrome with 8-year follow-up.a case of gorham disease with several years of follow-up is reported. at birth he had a mass in the thigh which was had pathology demonstrating a lymphangioma. by age 3 years, he had lymphedema of the ipsilateral foot and discrepant leg lengths. radiography revealed heterogenous dystrophy of the bones and osteolysis of the hallux. at age 6, a spontaneous fracture of the right tibia was treated with surgery. histopathology of a bone sample demonstrated bone remodelling, fibrous tissue, and large ...201622380698
gorham-stout disease affecting both hands: stabilisation during biphosphonate treatment.the gorham-stout syndrome is a rare condition in which spontaneous, progressive resorption of bone occurs. the aetiology is poorly understood. we report a patient with osteolysis of the metacarpal bones in both hands due to an increased number of stimulated osteoclasts. this suggests that early potent antiresorptive therapy with bisphosphonates may prevent local progressive osteolysis.201122379445
the properties of terrestrial laser system intensity for measuring leaf geometries: a case study with conference pear trees (pyrus communis).light detection and ranging (lidar) technology can be a valuable tool for describing and quantifying vegetation structure. however, because of their size, extraction of leaf geometries remains complicated. in this study, the intensity data produced by the terrestrial laser system (tls) faro ls880 is corrected for the distance effect and its relationship with the angle of incidence between the laser beam and the surface of the leaf of a conference pear tree (pyrus commmunis) is established. the r ...201122319374
development of a transgenic early flowering pear (pyrus communis l.) genotype by rnai silencing of pctfl1-1 and pctfl1-2.trees require a long maturation period, known as juvenile phase, before they can reproduce, complicating their genetic improvement as compared to annual plants. 'spadona', one of the most important european pear (pyrus communis l.) cultivars grown in israel, has a very long juvenile period, up to 14 years, making breeding programs extremely slow. progress in understanding the molecular basis of the transition to flowering has revealed genes that accelerate reproductive development when ectopical ...201222203321
gorham's disease of femur.gorham's disease is a rare condition of bones characterized by spontaneous massive and progressive osteolysis. less than 200 cases have been reported so far. femur is an uncommon site of gorham disease. we report a young female, presenting as pathological fracture of the femur, wherein rapid osteolysis of femur occurred. the clinical, pathological, and radiological picture suggested a diagnosis of gorham's disease. the patient was subjected to radiotherapy (40 gys) and put on bisphosphonates. at ...201122144753
new species, new records and new morphological characters of the genus tillicera spinola from china (coleoptera, cleridae, clerinae).two new species of the genus tillicera spinola, 1841 from china are described and illustrated: tillicera sensibilissp. n. from yunnan (also from myanmar, thailand and laos) and tillicera weniisp. n. from taiwan. tillicera bibalteata gorham, 1892, tillicera hirsuta (pic, 1926) and tillicera michaeli gerstmeier & bernhard, 2010 are newly recorded from china. tillicera auratofasciata (pic, 1927) is newly recorded in some provinces of china. a key to species of the genus from china is provided. rela ...201121998525
discovery of the female of pyrocoelia prolongata in taiwan (coleoptera, lampyridae).the female of pyrocoelia prolongata jeng & lai, a diurnal lampyrid species from taiwan, is described for the first time. a single individual was found in a small, shady, dry streambed at the edge of a mixed forest at 2700 m elevation. the individual glowed in darkness and would move its abdomen up and down when disturbed and as a deterring behavior. a key to the females of the species of pyrocoelia gorham in taiwan is provided. the morphology of photogenic organs of females and the function of b ...201121998501
the diagnosis and management of patients with idiopathic osteolysis.idiopathic osteolysis or disappearing bone disease is a condition characterized by the spontaneous onset of rapid destruction and resorption of a single bone or multiple bones. disappearing bone disorder is a disease of several diagnostic types. we are presenting three patients with osteolysis who have different underlying pathological features. detailed phenotypic assessment, radiologic and ct scanning, and histological and genetic testing were the baseline diagnostic tools utilized for diagnos ...201121995273
compositional changes in 'bartlett' pear ( pyrus communis l.) cell wall polysaccharides as affected by sunlight conditions.preharvest conditions can have a great impact on fruit quality attributes and postharvest responses. firmness is an important quality attribute in pear, and excessive softening increases susceptibility to bruising and decay, thus limiting fruit postharvest life. textural characteristics of fruits are determined at least in part by cell wall structure and disassembly. few studies have analyzed the influence of fruit preharvest environment in softening, cell wall composition, and degradation. in t ...201121980919
progressively bilateral resorption of the mandible.we describe a case of arrested spontaneous mandibular resorption for which there was no effective treatment. a 32-year-old male patient presented to our department with mandibular resorption so severe that the residual mandibular body bone resembled chopsticks. a provisional diagnosis of gorham-stout syndrome (gss) was made. vegf levels found in plasma and tissue fluid was believed to be a possible marker for the condition. although there was no histological evidence for the diagnosis, a combina ...201221925893
[polymorphism of microsatellite loci in cultivars and species of pear (pyrus l.)].using five ssr markers, polymorphism ofmicrosatellite loci was examined in 46 cultivars and five species of pear (pyrus ussuriensis, p. bretscgneideri, p. pyraster, and p. elaegnifolia). most of the accessions examined demonstrated the presence of unique allele sets. the degree of relationship between russian and western european pear cultivar was established. it was demonstrated that p. ussuriensis and its first generation progeny were genetically distant from typical cultivars of p. communis, ...201121786670
[gorham-stout syndrome--a case report]. 201321725909
how to prevent ripening blockage in 1-mcp-treated 'conference' pears.some european pear varieties treated with 1-methylcyclopropene (1-mcp) often remain 'evergreen', meaning that their ripening process is blocked and does not resume after removal from cold storage. in this work this was confirmed also to be the case in 'conference' pears. to reverse the blockage of ripening 1-mcp treatments combined with external exogenous ethylene were tested.201121681757
intraosseous csf fistula in a patient with gorham disease resulting in intracranial hypotension.a 7-year-old girl with a history of headaches and gorham disease was surgically treated in infancy for chiari i malformation. subsequent investigation revealed that her cerebellar tonsillar ectopia was due to a long-standing spinal csf-lymphatic fistula causing intracranial hypotension. percutaneous fistula closure was performed several times, resulting in transient symptomatic improvement.201121659480
a novel treatment approach for paediatric gorham-stout syndrome with chylothorax.to expand the treatment options in paediatric gorham-stout syndrome (gss) when conventional therapy is ineffective.201121605166
a case report of gorham-stout syndrome remission. 201221598027
definition and revision of the orthrius-group of genera (coleoptera, cleridae, clerinae).an "orthrius-group" of genera is proposed, and defined to include aphelochroa quedenfeldt, 1885; caridopus schenkling, 1908; dozocolletus chevrolat, 1842; gyponyx gorham, 1883; languropilus pic, 1940; orthrius gorham, 1876; pieleus pic, 1940; xenorthrius gorham, 1892; plus three new genera neorthriusgen. n., nonalatusgen. n. and pseudoastigmusgen. n. a phylogeny of the 11 constituent orthrius-group genera (analysis of 22 morphological characters using clerus geoffroy as the out-group taxon was p ...201121594111
carboxymethyl cellulose coating and low-dose gamma irradiation improves storage quality and shelf life of pear (pyrus communis l., cv. bartlett/william).carboxymethyl cellulose (cmc) coatings alone and in combination with gamma irradiation were tested for maintaining the storage quality and extending shelf life of pear. matured green pears were cmc coated at levels 0.25% to 1.0% w/v and gamma irradiated at 1.5 kgy. the treated fruit including control was stored under ambient (temperature 25 ± 2 °c, rh 70%) and refrigerated (temperature 3 ± 1 °c, rh 80%) conditions. irradiation alone at 1.5 kgy gave 8 and 4 d extension in shelf life of pear follo ...201621535615
symmetric metastatic melanoma of unknown primary, presenting as gorham-stout syndrome. 201621512676
differential expression of proteins in red pear following fruit bagging treatment.fruit bagging is a very effective method for study of fruit qualities and anthocyanin synthesis. the characterization of differentially expressed proteins that were isolated from both bagged and normal fruit skin tissue is apparently an essential parameter for understanding the effect of shading on fruit qualities and to understand the mechanism of fruit coloring in pyrus communis. proteome maps of both bagged and normal p. communis 'placer' fruit skin were obtained by performing two-dimensional ...201121409615
radiation therapy for gorham-stout syndrome: results of a national patterns-of-care study and literature review.the german cooperative group on radiotherapy for benign diseases conducted a national patterns-of-care study to investigate the value of radiation therapy (rt) in the management of gorham-stout syndrome.201121345608
gorham-stout syndrome of the pelvic girdle treated by radiation therapy: a case report.the gorham-stout syndrome (gss) is a rare, benign idiopathic and progressive disorder causing massive osteolysis due to a vascular hyperproliferation replacing the bony structure. clinical experience concerning the efficacy of radiation therapy (rt) is limited to about 50 of an overall 200 cases reported worldwide.201121336714
report of a rare case of gorham-stout disease of both shoulders: bisphosphonate treatment and shoulder replacement.massive osteolysis known as gorham-stout disease is a rare idiopathic disorder typically affecting long bones in a unifocal pattern. angiomatosis is strongly connected to the osteolysis. weather angiomatosis is the cause or the result of osteolysis is subject of intense discussion (kawasaki et al. (2003), möller et al. (1999), radhakrishnan and rockson (2008)). there are about 200 cases described since 1955. our patient is a 77-year-old female patient with osteolyses of both shoulders involving ...201122937447
gorham-stout syndrome with chylothorax in a six-year-old boy.gorham-stout syndrome, also called "disappearing bone disease, diffuse cystic angiomatosis of bone, disseminated lymphangiomatosis, gorham's vanishing bone disease, phantom bone disease or idiopathic massive osteolysis, is a rare disease of unknown etiology and pathogenesis. it is characterized by rapidly progressive localized massive osteolysis associated with proliferation of vascular structures of benign origin in which the absence of new bone formation is representative. when it is complicat ...201121188554
orbital roof encephalocele mimicking a destructive neoplasm.the purpose of this case report is to report an orbital roof encephalocele mimicking a destructive orbital neoplasm. orbital roof encephalocele is uncommon but can mimic neoplasm. one potential mechanism for the orbital roof destruction is a post-traumatic "growing orbital roof fracture." the growing fracture has been reported mostly in children but can occur in adults. alternative potential etiologies for the encephalocele are discussed, including gorham syndrome. orbital roof encephalocele is ...201121178797
chronic recurrent gorham-stout syndrome with cutaneous involvement.type iv osteolysis or gorham-stout syndrome is a rare condition characterized by recurrent vascular tumors that disrupt normal anatomical architecture. gorham-stout syndrome is most commonly associated with the skeletal system with resulting replacement of bone with scar tissue following tumor regression. the loss of entire bones has given gorham-stout syndrome the moniker vanishing bone disease. natural progression of gorham-stout syndrome is characterized by spontaneous disease resolution. how ...201021139956
anesthetic management of a pregnant woman with gorham-stout disease.gorham-stout disease is a rare disorder of bone loss and proliferation of lymphatic and vascular tissue (lymphangiomatosis). a 30-year-old nulliparous woman with gorham-stout disease presented at 8weeks of gestation with a fused cervical spine. at 31weeks she developed basilar invagination and neurological symptoms that were managed with a neck brace. anesthetic considerations were those of airway compromise, development of severe preeclampsia and kasabach-merritt coagulopathy. elective tracheos ...201121111606
could local deliver of bisphosphonates be a new therapeutic choice for gorham-stout syndrome?the gorham-stout syndrome is a rare condition in which spontaneous, progressive resorption of bone occurs. even though the prognosis of the condition is generally considered to be good, gorham-stout syndrome can cause severe debilitation. in approximately 13% of recorded cases, death ensues. the treatment modalities for gorham-stout syndrome include surgery, radiation therapy, anti-osteoclastic medications, however there is no known successful treatment. to date, the etiology of gorham-stout syn ...201121044823
gorham-stout syndrome: phantom bone disease.gorham-stout syndrome is a disease that presents idiopathic osteolysis of a bone or closely contiguous area. the etiology is unknown. it is a rare condition that is difficult to diagnose, and its treatment is controversial. it affects individuals irrespective of age or sex. in this study, we conducted a bibliographic review of the disease, specifically focusing on the differential diagnosis, and we demonstrated the follow-up on a patient with this syndrome from the time of its diagnosis, through ...201727026974
gorham-stout disease: the experience of the rizzoli institute and review of the literature.gorham-stout disease (also known as "disappearing bone disease") was first described by jackson in 1838, but was properly defined by gorham and stout in a series of 24 patients in 1954-1955. it is a rare disease of unknown etiology (about 200 cases reported in the literature) characterized by spontaneous progressive resorption of bone without malignant proliferation of vascular structures. the diagnosis is one of exclusion and it is based on combined histological, radiological, and clinical feat ...201120972870
treatment of gorham-stout disease with zoledronic acid and interferon-α: a case report and literature review.gorham-stout disease is a rare disease characterized by osteolysis, angiomatosis, and soft-tissue swelling. it is a diagnosis of exclusion and has an unknown etiology. chylothorax is a common complication of the disease that is associated with a high mortality rate. there is no standard of treatment. we report a case of a 16-year-old female with gorham-stout disease and recurrent pleural effusions who was successfully treated with concurrent zoledronic acid and peg-interferon α-2b.201020962674
mapping of an anthocyanin-regulating myb transcription factor and its expression in red and green pear, pyrus communis.'max red bartlett' is a red bud mutation of the yellow pear (pyrus communis l.) cultivar 'williams' (known as 'bartlett' in north america). anthocyanins are the most important pigments for red colour in fruits. synthesis of anthocyanins is mediated by a number of well-characterized enzymes that include chalcone synthase (chs), flavanone-3-hydroxylase (f3h), dihydroflavonol-4-reductase (dfr), anthocyanidin synthase (ans), and udp-glucose:flavonoid-3-o-glucosyltransferase (ufgt). expression of the ...201020951056
teaching neuroimages: vanishing calvarium in gorham disease. 201020938023
gorham-stout disease.gorham-stout disease is a rare disease of unknown etiology. it is characterized by spontaneous excessive replacement of bone by proliferative non-neoplastic thin-walled lymphatic and/or blood vessels. histology shows positive stain for the lymphatic endothelial marker lyve-1 (lymphatic vascular endothelial hyaluronan receptor-1) and many lymphatic growth factors (pdgf-bb, vegf-c, vegfr-3). patients may present with localized pain and/or weakness and radiographic evidence of massive osteolysis in ...201020727303
a rare cause of dyspnea due to chylothorax.we report the case of an 8-year-old boy who presented to the emergency department of another hospital and was referred to our pediatric intensive care unit with dyspnea and tachypnea of recent onset. the diagnosis of massive chylothorax with mediastinal shift was made on chest radiographs and ct scan. initial investigations revealed no definite cause for the chylothorax. on later radiography and ct imaging with 3d surface rendering, a marked bone loss of the left ribs, clavicle, and shoulder joi ...201020721678
[gorham-stout syndrome (gss) with fulminant aseptic osteonecrosis of the shoulder].we report here a case of the rare gorham-stout syndrome (gss) of the humerus. gss is a disease in the course of which spontaneous idiopathic osteolysis occurs. we describe a case of a 46-year-old business economist who suffered an inadequate trauma and within 3 weeks developed self-limited idiopathic osteolysis of the left humerus head, also affecting the rotator cuff. this could be diagnosed especially on the basis of histopathological findings. hereupon we indicated operative repair by means o ...201020714707
two neutral variants segregating at the gametophytic self-incompatibility locus of european pear (pyrus communis l.) (rosaceae, pyrinae).extensive survey of the s-locus diversity of plant species with rnase-based gametophytic self-incompatibility has failed to identify neutral variation segregating within s-allele specificities. although this is the expected result according to population genetics theory, it conflicts with recent models of s-allele evolution, which suggest that new specificities might arise by a continuous process of subtle changes that individually do not alter the specificity of the s-genes, but whose cumulativ ...201020701704
first report of effective and feasible treatment of multifocal lymphangiomatosis (gorham-stout) with bevacizumab in a child. 201020605931
thirty-year follow-up of patient with gorham disease (massive osteolysis) treated with hip arthroplasty.gorham disease (also known as massive osteolysis or disappearing bone disease) is an exceedingly rare, peculiar entity of uncertain cause and unpredictable prognosis. there is no proven mode of therapy or consensus on treatment. we present 30-year follow-up on a patient successfully treated with resection, total hip arthroplasty, and radiation with no recurrence of disease. our observations in this case suggest that surgical treatment and reconstruction can lead to good long-term function and di ...201120580190
survey of susceptibility to abamectin of pear psylla (hemiptera: psyllidae) in northern italy.the pear psylla, cacopsylla pyri l. (hemiptera: psyllidae), is a relevant pest of pear, pyrus communis l., trees in emilia-romagna region (northern italy). the susceptibility to the insecticide abamectin was evaluated at different times of the year on c. pyri populations undergoing different control strategies within conventional, integrated, and organic farms. the tests performed were the egg spray and the topic and dip bioassay on adults. the larval mortality was evaluated by dip bioassay on t ...201020568628
gorham-stout syndrome of the petrous apex causing chronic cerebrospinal fluid leak.to describe the clinical course, diagnostic features, and treatment of a case of gorham-stout syndrome involving the petrous apex and causing chronic cerebrospinal fluid (csf) leak.201020502378
cardiovascular images. chylotamponade: an unusual manifestation of gorham-stout syndrome. 201020233864
massive cranial osteolysis, skin changes, growth retardation and developmental delay: gorham syndrome with systemic manifestations?we report on a 16-month-old girl with multiple swellings on her skull due to massive osteolysis, growth retardation, facial anomalies, and wrinkly skin with mosaic hypopigmentation. she also had severe hypercalcemia, which gradually returned to normal levels. the condition likely represents gorham syndrome with systemic manifestations.201020186788
gorham-stout disease of the temporal bone.gorham-stout (gs) disease is a rare disease of the bone and is also known as massive osteolysis. less than 200 cases have been reported in the world literature. a 29-year-old female with a diagnosis of gs disease was identified. she complained of aural fullness and tinnitus bilaterally. demineralization and moth-eaten changes of the osseous structures of the skull base and posterior fossa were prominent. the left mastoid air cells were opacified and erosion extended to the left jugular foramen, ...201020131367
commentary: massive osteolysis or gorham-stout disease. 201020123408
[gorham syndrome, a rare disease...]. 201020087270
use of zoledronic acid in the treatment of gorham's disease.gorham's disease (gorham-stout syndrome) is a rare condition of unknown etiology involving a localized endothelial proliferation of lymph vessels resulting in destruction with bone resorption. the syndrome is rarely seen in the facial skeleton and has a large variety of prognoses and treatments. a case of this syndrome in a 9-year-old boy is presented. the clinical aspects are described, together with the treatment involving zoledronic acid. other treatments described in the literature are revie ...201020074817
conservative treatment of a pathologic fracture in a patient with gorham-stout disease.gorham-stout disease, or massive osteolysis, is an extremely rare idiopathic condition characterized by spontaneous, localized relentless resorption of one or several contiguous bones. there is no known successful treatment. autologous bone graft also resorbs. it is a condition with difficult diagnosis, treatment, and prognosis. we report an extreme case of massive osteolysis of the maxillofacial complex. unique to this case is that the patient presented with a mandibular fracture, and was takin ...201020034818
compatible and self-incompatible pollination in pyrus communis displays different polyamine levels and transglutaminase activity.the polyamine (pa) content and the transglutaminase (tgase) activity have been investigated in pyrus communis pollination with compatible and self-incompatible (si) pollen in order to deepen their possible involvement in the progamic phase of plant reproduction. the pa distribution as free, perchloric acid (pca)-soluble and pca-insoluble fractions in ungerminated (ugp), germinating pollen (gp), styles and pollinated styles with compatible and si pollens is discussed in the light of a possible ro ...201020020163
radiological and ct findings in a extensive upper-limb involvement of gorham's disease: a case report.the gorham-stout syndrome (gorham's massive osteolysis) is a rare condition in which spontaneous, progressive resorption of bone occurs. the etiology is poorly understood.200919829979
spermidine levels are implicated in heavy metal tolerance in a spermidine synthase overexpressing transgenic european pear by exerting antioxidant activities.to verify whether spermidine synthase (spds) can confer long-term multi-heavy metal tolerance, in vitro shoots of a transgenic european pear (pyrus communis l. 'ballad') line #32 overexpressing apple spds (mdspds1), as well as a wild type (wt) line, were subjected to stress using either cdcl(2), pbcl(2), zncl(2), or a combination thereof. based on either shoot height increment or fresh weight and morphological changes upon heavy metal stress, the performance of the transgenic line #32 was better ...201019544002
[clinical features of gorham-stout syndrome and literature review].to describe the clinical presentations, radiographic findings and histological pathology of bones, diagnosis, treatment options and prognosis for patients with gorham-stout syndrome (gss).200919484972
gorham-stout syndrome with chylothorax: successful remission by interferon alpha-2b.gorham-stout syndrome is a rare disease and most often recognized in children and young adults. chylothorax is a serious complication of gorham-stout syndrome. the treatment of either gorham-stout syndrome or chylothorax is still a dilemma. we described a 9-year-old girl with gorham-stout syndrome and chylothorax who was admitted to our hospital because of dyspnea, orthopnea, and bone pain, and treated with interferon alpha-2b.200919434689
gorhams disease: vanishing bone syndrome.gorhams disease, also known as massive osteolysis or vanishing bone disease is an extremely rare bone disease. it is characterized by angiomatosis with adjacent bone resorption. we report an 8-years old boy with the disease who was managed successfully with alpha 2b interferon therapy.200919346574
vanishing bone disease: review and case reports.vanishing bone disease is a rare idiopathic disease, leading to extensive loss of bony matrix, replaced by proliferating thin-walled vascular channels and fibrous tissue. there are >191 cases reported in the english literature. gorham and stout made the first overview of the disease in 1955 and they first presented 24 cases known at that time. the etiology remains speculative, the prognosis unpredictable, and effective therapy still unknown. the disease can be monostotic or polyostotic although ...200819292231
progressive gorham disease of the forearm.gorham's-stout disease is a rare but potentially debilitating disease consisting of massive bone osteolysis and bone resorption associated with vascular proliferation and increased osteoclastic activity. although it can present in a wide variety of forms, it typically involves bones formed by intramembranous ossification such as the skull, pelvis, and scapula. it can occur spontaneously or after trauma. most cases are monofocal and resolved spontaneously, although there are reports of multifocal ...200819292225
the gorham-stout syndrome: one lung ventilation with a bronchial blocker. a case of gorham's disease with chylothorax. 200919207918
microscale mechanisms of gas exchange in fruit tissue.* gas-filled intercellular spaces are considered the predominant pathways for gas transport through bulky plant organs such as fruit. here, we introduce a methodology that combines a geometrical model of the tissue microstructure with mathematical equations to describe gas exchange mechanisms involved in fruit respiration. * pear (pyrus communis) was chosen as a model system. the two-dimensional microstructure of cortex tissue was modelled based on light microscopy images. the transport of o(2) ...200919192195
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