Archive of Medical Research

SAPHO syndrome with rapidly progressing destructive spondylitis: two cases treated surgically.

The authors present two cases of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome with rapidly progressing destructive spondylitis treated surgically. The spinal lesions in SAPHO syndrome generally have a good prognosis and rarely cause the structural destruction or neurological deterioration. Case 1: a 63-year-old female had palmoplantar pustulosis for 2 years. At first, she only felt a pain in the nape with no inducing factor. Two months later, she had incomplete quadriplegia (ASIA scale C). Magnetic resonance imaging showed destruction of C4-C7, kyphotic deformity, and severe compression of the spinal cord. Decompression and reconstruction surgery using anterior and posterior approach improved her paralysis. Case 2: a 69-year-old female complained of persistent back pain. Magnetic resonance imaging revealed spondylitis of T7-T9. Although there were no typical skin lesions, we diagnosed SAPHO syndrome by hyperostosis of the sternocostoclavicular joint and sacral joint. Destruction with kyphotic deformity of the spine progressed gradually for 3 months. Curettage and reconstruction surgery using thoracic endoscope relieved her pain and prevented the destruction of the spine. The histopathology of the specimen obtained surgically showed non-specific inflammation in both cases. Spondylitis in SAPHO syndrome may cause severe destruction and kyphotic deformity followed by paralysis.

Takigawa T, Tanaka M, Nakahara S, Sugimoto Y, Ozaki T.

Department of Orthopaedic Surgery, Okayama University Hospital, 2-5-1 Shikata-cho, Okayama City, Okayama, 700-8558, Japan, takigawa2004@yahoo.co.jp.

Spores of the mycorrhizal fungus Glomus mosseae host yeasts that solubilize phosphate and accumulate polyphosphates.

The present paper reports the presence of bacteria and yeasts tightly associated with spores of an isolate of Glomus mosseae. Healthy spores were surface disinfected by combining chloramine-T 5%, Tween-40, and cephalexin 2.5 g L(-1) (CTCf). Macerates of these spores were incubated on agar media, microorganisms were isolated, and two yeasts were characterized (EndoGm1, EndoGm11). Both yeasts were able to solubilize low-soluble P sources (Ca and Fe phosphates) and accumulate polyphosphates (polyPs). Sequence analysis of 18S ribosomal deoxyribonucleic acid showed that the yeasts belong to the genera Rhodotorula or Rhodosporidium (EndoGm1) and Cryptococcus (EndoGm11). Results from inoculation experiments showed an effect of the spore-associated yeasts on the root growth of rice, suggesting potential tripartite interactions with mycorrhizal fungi and plants.

Mirabal Alonso L, Kleiner D, Ortega E.

National Institute of Agricultural Sciences (INCA), Havana, Cuba.

Childhood chronic kidney disease in a developing country.

We have retrospectively reviewed the records of children aged >1 month to 16 years who had been referred to the Department of Pediatrics of Prince of Songkla University\’s Faculty of Medicine, a tertiary referral center in Thailand, between 1982 and 2005 and subsequently diagnosed with chronic kidney disease (CKD). Our aim was to evaluate the prevalence and etiology of CKD in southern Thailand. There were 101 cases of CKD, with one case each diagnosed in 1988, 1989 and 1993, respectively, and 98 cases diagnosed between 1994 and 2005. These latter cases were divided into two 6-year periods: an early period (1994-1999), with 32 cases, and a later period (2000-2005), with 66 cases. The majority of this pediatric population with CKD were male (62/101, 61.4%). The etiologies of CKD were 35 cases of chronic glomerulonephritis (CGN) (34.7%), 29 of genitourinary tract (GU) anomalies (28.7%), nine of systemic lupus erythematosus (SLE) (8.9%), four malignancies (4.0%), four miscellaneous (4.0%) and 19 of unknown causes (18.8%). The patients were divided into age groups of <2 years (20 CKD patients), 2-6 years (15), >6-10 years (22), >10-13 years (20) and >13 years (24). The etiologies of CKD were significantly different in each age group, with GU anomalies and glomerulonephritis being the major causes of CKD in children aged </=6 years (20/36, 55.6%) and >6 years (40/65, 61.5%), respectively. In conclusion, the incidence of CKD in our university hospital situation was not rare, with the prevalence doubling during the past 6 years, and the etiologies varying by age group.

Vachvanichsanong P, Dissaneewate P, McNeil E.

Department of Pediatrics, Faculty of Medicine, Prince of Songkla University, Hat Yai, 90110, Thailand, vprayong@msn.com.

Outcome after renal transplantation. Part I: Intellectual and motor performance.

Comprehensive information on neurodevelopmental outcome in children and adolescents with chronic kidney disease is still limited. Intellectual performance (IP) and motor performance (MP) were systematically assessed in 27 patients at a median age of 14.1 years (range 6.5-17) and 6 years (range 0.5-12.7) after renal transplantation (RTPL). IP was analyzed with the Wechsler Intelligence Scale for Children-III (WISC-III) in 25 patients and by the Kaufman Assessment Battery for Children in two patients. MP was evaluated by the Zurich Neuromotor Assessment. Median full- scale intelligent quotient (FSIQ) was 97 (range 49-133). Twenty-one patients had an FSIQ >/= 85 (i.e. >/= mean-1 standard deviation). The five patients with neurological comorbidity had a median FSIQ of 81 (range 49-101). Verbal IQ (VIQ) (median 104; range 50-146) was significantly (p < 0.01) higher than performance IQ (PIQ) (median 88; range 48-117). The PIQ was significantly lower compared with controls (p < 0.007), and patients scored significantly lower compared with controls in five of 11 subtests of the Wechsler Scale. All MP tasks were significantly (p < 0.01) lower than in controls, and also in children without neurological comorbidity. Socioeconomic status was significantly correlated with FSIQ (p = 0.03). IP after RTPL was within the normal range for the majority of children. PIQ was lower compared with VIQ, and MP was significantly affected in all children after RTPL.

Falger J, Latal B, Landolt MA, Lehmann P, Neuhaus TJ, Laube GF.

Nephrology Unit, University Children’s Hospital, Steinwiesstrasse 75, 8032, Zurich, Switzerland.

Leishmaniasis treatment-a challenge that remains: a review.

Leishmaniasis is a disease caused by flagellate protozoan Leishmania spp. and represents an emergent illness with high morbidity and mortality in the tropics and subtropics. Since the discovery of the first drugs for Leishmaniasis treatment (i.e., pentavalent antimonials), until the current days, the search for substances with antileishmanial activity, without toxic effects, and able to overcome the emergence of drug resistant strains still remains as the current goal. This article reports the development of new chemotherapies through the rational design of new drugs, the use of products derived from microorganisms and plants, and treatments related to immunity as new alternatives for the chemotherapy of leishmaniasis.

Santos DO, Coutinho CE, Madeira MF, Bottino CG, Vieira RT, Nascimento SB, Bernardino A, Bourguignon SC, Corte-Real S, Pinho RT, Rodrigues CR, Castro HC.

Laboratório de Biopatógenos e Ativação Celular (LaBiopAc), Departamento de Biologia Celular e Molecular, Instituto de Biologia, Universidade Federal Fluminense (UFF), Av. Barros-Terra s/n Valonguinho, Niterói, Rio de Janeiro, CEP 24001-970, Brazil, santosdilvani@gmail.com.

Field studies indicating reduced activity of ivermectin on small strongyles in horses on a farm in Central Kentucky.

Field studies (n = 6) were completed on evaluation of activity of ivermectin (200 mug/kg) paste formulation against small strongyles in horses (foals, yearlings, and older animals) on a farm (Farm MC) in Central Kentucky in late 2006 and during 2007. A girth tape was used to estimate body weights which were then used to calculate the proper dose rate of ivermectin. The foals, yearlings, and some of the older horses were born and raised on the farm. However, most of the older horses which were not raised on the farm had been there for several years. The horse herd was given ivermectin exclusively, usually four times a year, since 1990. An exception was that during the foal\’s period of life fenbendazole, pyrantel pamoate, and oxibendazole were given occasionally besides ivermectin. Efficacy of drug activity was determined by pretreatment and posttreatment counts of strongyle eggs per gram of feces (EPGs). Culture of strongyle eggs in feces from some of the horses showed that only small strongyle larvae were present. The research included two studies (A and B) in foals (n = 24) and four studies (C, D, E, and F) in yearlings (n = 13) alone or with older horses (n = 10). For each of the studies (B through F), there was a treated and a nontreated group. These groups were switched for each treatment, i.e., the treated group in one study was the nontreated group in the next study and vice versa. Eggs per gram of feces counts were determined at 1- or 2-week posttreatment intervals for 4 weeks for study A and 6 weeks for studies B through F. Also, for studies B, E, and F, counts of EPGs were done either two or three times during the third week posttreatment. The studies showed a similar posttreatment pattern of strongyle EPG counts beginning to return at about 4 weeks and increasing at 5 and 6 weeks posttreatment. Two horses in study E and one in study F had low EPG values toward the end of the third week posttreatment. The results of this ivermectin investigation showed that the strongyle EPG counts started returning about twice as quickly post-ivermectin-treatment of horses than when the drug was first marketed in the early 1980s.

Lyons ET, Tolliver SC, Ionita M, Lewellen A, Collins SS.

Department of Veterinary Science, Gluck Equine Research Center, University of Kentucky, Lexington, KY, 4056-0099, USA, elyons1@uky.edu.

Polymorphisms in the novel serotonin receptor subunit gene HTR3C show different risks for acute chemotherapy-induced vomiting after anthracycline chemotherapy.

The aim of this study was to correlate chemotherapy-induced nausea and vomiting (CINV) with commonly occurring single nucleotide polymorphisms (SNP) in the 5-hydroxytryptamine receptor 3 genes (HTR3). Women with breast cancer without previous chemotherapy were eligible for this prospective study. All patients received epirubicin, with or without cyclophosphamide, and preventive medication with ondansetron and dexamethasone. The patients documented every vomiting event on an hourly basis. Real-time polymerase chain reaction (PCR) analysis was performed for the following nonsynonymous SNPs: p.Y129S (HTR3B), p.K163N (HTR3C) and p.A405G (HTR3C). The overall proportion of patients (total n = 110) who reported vomiting in the first 24 h after chemotherapy was 31.8%. The variant genotype of K163N (HTR3C) was associated with vomiting, which occurred in 50.0% (P = 0.009). Polymorphisms in the HTR3C gene could serve as a predictive factor for CINV in patients undergoing moderately emetogenic chemotherapy.

Fasching PA, Kollmannsberger B, Strissel PL, Niesler B, Engel J, Kreis H, Lux MP, Weihbrecht S, Lausen B, Bani MR, Beckmann MW, Strick R.

University Breast Center Franconia, Erlangen University Hospital, Universitaetsstrasse 21-23, 91054, Erlangen, Bavaria, Germany, peter.fasching@gyn.med.uni-erlangen.de.

What\’s new in cystic fibrosis? From treating symptoms to correction of the basic defect.

Chronic relentless lung infection and pancreatic insufficiency are the cardinal features of cystic fibrosis (CF), a life-shortening autosomal recessive disease. Mutations in the \’cystic fibrosis transmembrane conductance regulator\’ (CFTR) are currently classified into five groups according to their repercussion on CFTR protein synthesis and its chloride channel function. Stop codon mutations (class I) result in a truncated nonfunctional CFTR, class II mutations consist of aberrantly folded CFTR protein that is degraded by the cell quality control system, while class III mutations lead to defective regulation of the CFTR protein and, consequently, the absence of CFTR function. These three classes usually lead to a classic CF phenotype with pancreatic insufficiency. CFTR mutations that lead to defective chloride conductance are grouped together in class IV. Class V mutations interfere with normal transcription, thereby reducing the amount of otherwise normal CFTR. These latter two classes are mostly associated with a milder expression of the disease. In the absence of CFTR function, unrestrained Na+ absorption and the failure of active Cl- secretion lead to a decreased airway surface liquid (ASL) volume and subsequent failure of normal mucociliary clearance. This review highlights recent therapeutic strategies that either target the underlying defect or the early steps in CF pathophysiology. To date, gene therapy has failed to demonstrate a clinical benefit after repeated administration. Mutation-specific chloride channel correction pharmacotherapy is currently being developed, an example of which is PTC124, a new chemical compound that selectively induces read-through of premature stop codons. However, clinical efficacy for most of the compounds still has to be proven in large clinical trials. The positive effect of nebulised hypertonic saline on mucociliary clearance is based on the restoration of ASL height. Recent advances in the current treatment of lung infection and inflammation are highlighted in this review. Lung transplantation should be considered in terminally ill patients, but the timing of the transplantation is crucial: transplanting too early shortens survival, while transplanting too late results in patients dying on the waiting list.

Proesmans M, Vermeulen F, De Boeck K.

Department of Pediatrics, University Hospital of Leuven, Herestraat 49, 3000, Leuven, Belgium, marijke.proesmans@uzleuven.be.

The significance of lobular neoplasia on needle core biopsy of the breast.

The management of a core biopsy diagnosis of lobular neoplasia is controversial. Detailed radiological-pathological review of 47 patients with cores showing classical lobular neoplasia was performed (patients with pleomorphic lobular carcinoma in situ (LCIS) or associated risk lesions were considered separately). Immediate surgical excision in 25 patients showed invasive carcinoma in 7, ductal carcinoma in situ (DCIS) in 1 and pleomorphic LCIS in 1; radiological-pathological review showed that the core biopsy missed a mass in 5, missed calcification in 2 and that calcification appeared adequately sampled in 2. Nineteen patients had follow-up of at least 2 years. Four patients developed malignancy at the site of the core biopsy (invasive carcinoma in three, DCIS in one); one carcinoma was mammographically occult, one patient had dense original mammograms and two had calcifications apparently adequately sampled by the core. In conclusion, most carcinomas identified at the site of core biopsy showing lobular neoplasia were the result of the core missing the radiological lesion, emphasising the importance of multidisciplinary review and investigation of any discordance. Some carcinomas were found after apparently adequate core biopsy, raising the question of whether excision biopsy should be considered after all core biopsy diagnoses of lobular neoplasia.

Menon S, Porter GJ, Evans AJ, Ellis IO, Elston CW, Hodi Z, Lee AH.

Department of Histopathology, Nottingham University Hospital, City Hospital Campus, Hucknall Road, Nottingham, NG5 1PB, UK.

Voltage-gated calcium channels in chronic pain: emerging role of alternative splicing.

N- and T-type voltage-gated calcium channels are key established players in chronic pain. Current work suggests that alternative splicing of these channels constitutes an important aspect in the investigation of their roles in the pathogenesis of chronic pain. Recent N-type channel studies describe a nociceptor-enriched alternatively spliced module responsible for voltage-independent G protein modulation and internalization, which is implicated in the control of distinct nociceptive pathways. On the contrary, although a large body of work has demonstrated that peripheral Ca(v)3.2-encoded T-type currents are involved in several types of chronic pain, little is known with respect to the expression of numerous newly discovered splice variants in specific pain pathways. The elucidation of the new layers of molecular complexity uncovered in N- and T-type channel splice variants and their respective locations and roles in different pain pathways will allow for the development of better therapeutic strategies for the treatment of chronic pain.

Swayne LA, Bourinet E.

Département de Physiologie, Institut de Génomique Fonctionnelle, 141 rue de la Cardonille, 34094, Montpellier, France.