Publications

Book Review - Scholarly Journals--Published

• Bauer M, Saldarriaga W, Wolfe SA, Beckwith JB, Frias JL, Cohen MM: "Two Extraordinarily Severe Cases of Treacher Collins Syndrome", Am J Med Genet. 161A:445-452, 2013. (02/2013)
• Beckwith JB: Book Review: Stocker and Dehner''s Pediatric Pathology, 3rd Edition. Pediatr Devel Pathol 14:429-30, 2001. (07/2011 - 06/2012)
•  Book Review: Beckwith JB: Arch. Pediatr. Adol. Med. (2008), 163(8),p.  796. Review of Graham JM: Smith''s Recognizable Patterns of Human Deformation, Edition 3 (2007).   (08/2008 - 08/2009)

Scholarly Journals--Published

• Beckwith JB: "Congenital Malformations: From Superstition to Understanding", Virchows Archiv 460:609-19, 2012. (08/2012)
• Gadd S, Huff V, Grundy PE, Ruteshouser EC, Huang C-C, jennings L, Breslow N, Green D, Dome JS, Beckwith JB, perlman EJ: "Clinically Relevant Subsets Identified by Gene Expression Patterns Support A Revised Ontogenic Model of Wilms Tumor: A Children''''s Oncology Group Study". Neoplasia 14:742-56, 2012 (07/2012)
• Lange J, Peterson SM, Takashima JR, Grigoriev Y, Ritchey ML, Shamberger RC, Beckwith JB, Perlman E, Green DM, Breslow NE: "Risk Factors for End Stage Renal Disease in Non-WT1-Syndromic Wilms Tumors" J. Urol 186: 378-86, 2001. (07/2011 - 06/2012)
•  Weksberg R, Shuman C, Beckwith JB: Beckwith-Wiedemann Syndrome. European Journal of Medical Genetics (2009, published online prior to print 24 June 2009.   (08/2008 - 08/2009)
•  Weksberg R, Shuman C, Beckwith JB: Beckwith-Wiedemann Syndrome. European Journal of Medical Genetics (2009) Published online 24 June 2009.   (08/2008 - 08/2009)
• Malogolowkin M, Cotton CA, Green DM, Breslow NE, Perlman E, Miser J, Ritchey ML, Thomas PR, Grundy PE, D''Angio GJ, Beckwith JB, Shamberger RC, Haase GM, Donaldson M, Weetman R, Coppes MJ, Shearer P, Coccia P, Kletzel M, Macklis R, Tomlinson G, Huff V, Newbury R, Weeks D: Treatment of Wilms tumor relapsing after initial treatment with vincristine and actinomycin D: A report from the National Wilms Tumor Study Group. Pediatr Blood Cancer 50:236-41, 2008.   (07/2008 - 07/2009)
• Vujanic GM, Kelsey A, Perlman EJ, Sandstedt B, Beckwith JB. "Anaplastic sarcoma of the kidney. A clinicopathological study of 20 cases of a new entity with polyphenotypic features.." Am J Surg Pathol 31:1459-1468 (Oct. 2007). Abstrac t on Pub Med (06/2007 - 06/2008)
• Beckwith JB. "A hitchhiker''s guide to the older literature of descriptive teratology." Am J Med Genet, Part A . Volume 143A: 2862-2867 (December, 2007). Abstract on Pub Med (06/2007 - 06/2008)
• Malagolowkin M, Cotton CA, Green DM, Beckwith JB et al. "Treatment of Wilms tumor relapsing after initial treatment with vincristine, actinomycin D, and doxorubicin. A report from the National Wilms Tumor Study Group." Pediatr Blood Cancer 50:236-241, (Feb.2008)  Abstract on Pub Med (06/2007 - 06/2008)
• Beckwith JB. "Management of incidentally encountered nephrogenic rests." J Pediatr Hematol Oncol 29.6 (2007): 353-354. Discusses management of apparent precursor lesions of Wilms tumor discovered incidentally during renal imaging studies for unrelated purposes. (06/2007)
• Ellison DA, Parham DM, Bridge J, Beckwith JB. "Immunohistochemistry of primary malignant neuroepithelial tumors of the kidney: a potential source of confusion? A study of 30 cases from the National Wilms Tumor Study Pathology Center." Hum Pathol 38.2 (2007): 205-211.  Abstract available on Pub Med (02/2007)
• Breslow NE, Beckwith JB, Perlman EJ, Reeve AE. "Age distributions, birth weights, nephrogenic rests, and hetrogeneity in the pathogenesis of Wilms tumor." Pediatr Blood Cancer 47.3 (2006): 260-267. (09/2006)
• Huang CC, Cutcliffe C, Coffin C, Sorensen PH, Beckwith JB, Perlman EJ. "Classification of malignant pediatric renal tumors by gene expression." Pediatr Blood Cancer 46.7 (2006): 728-738. BACKGROUND: The most common malignant renal tumors of childhood are Wilms tumor (WT), clear cell sarcoma of the kidney (CCSK), cellular mesoblastic nephroma (CMN), and rhabdoid tumor of the kidney (RTK). Because these tumors present significant diagnostic difficulties, the goal was to define diagnostically useful signatures based on gene expression. PROCEDURES: Gene expression analysis using oligonucleotide arrays was performed on a training set of 47 tumors (10 CCSKs, 9 CMNs, 8 RTKs, and 20 WTs). Classifiers were developed for each tumor type using variations of compound covariate class predictor. The classifiers were applied to an independent test set of 72 tumors (3 CMN, 7 CCSK, 4 RTK, and 58 WT). Central review diagnosis was utilized as the gold standard. Correlation with the institutional diagnosis and qualitative estimation of confidence levels at the time of central review were noted. RESULTS: Within the training set, classifiers resulted in no errors when >10 genes were utilized. Top genes in each classifier were verified using quantitative reverse transcription-polymerase chain reaction (RT-PCR). Applying the classifiers to the test set, 71 of 72 tumors were correctly classified with a confidence level of >99%. The exception was incorrectly classified by the gold standard. In comparison, by histopathology 31% of the non-WT were not accurately classified by the local institution, and 29% were classified with (06/2006)
• Dome JS, Cotton CA, Perlman FJ, Beckwith JB et al.. "Treatment of anaplastic histology Wilms tumor: results from the fifth National Wilms Tumor Study.." J Clin Oncol 24.15 (2006): 2352-2358. (05/2006)
• Dome JS, Cotton CA, Perlman EJ, Breslow NE, Kalapurakal JA, Ritchey ML, Grundy PE, Malogolowkin M, Beckwith JB, Shamberger RC, Haase GM, Coppes MJ, Coccia P, Kletzel M, Weetman RM, Donaldson M, Macklis RM, Green DM. "Treatment of anaplastic histology Wilms' tumor: results from the fifth National Wilms' Tumor Study.." J Clin Oncol 24.15 (2006): 2352-2358. PURPOSE: An objective of the fifth National Wilms' Tumor Study (NWTS-5) was to evaluate the efficacy of treatment regimens for anaplastic histology Wilms' tumor (AH). PATIENTS AND METHODS: Prospective single-arm studies were conducted. Patients with stage I AH were treated with vincristine and dactinomycin for 18 weeks. Patients with stages II to IV diffuse AH were treated with vincristine, doxorubicin, cyclophosphamide, and etoposide for 24 weeks plus flank/abdominal radiation. RESULTS: A total of 2,596 patients with Wilms' tumor were enrolled onto NWTS-5, of whom 281 (10.8%) had AH. Four-year event-free survival (EFS) and overall survival (OS) estimates for assessable patients with stage I AH (n = 29) were 69.5% (95% CI, 46.9 to 84.0) and 82.6% (95% CI, 63.1 to 92.4). In comparison, 4-year EFS and OS estimates for patients with stage I favorable histology (FH; n = 473) were 92.4% (95% CI, 89.5 to 94.5) and 98.3% (95% CI, 96.4 to 99.2). Four-year EFS estimates for patients who underwent immediate nephrectomy with stages II (n = 23), III (n = 43), and IV (n = 15) diffuse AH were 82.6% (95% CI, 60.1 to 93.1), 64.7% (95% CI, 48.3 to 77.7), and 33.3% (95% CI, 12.2 to 56.4), respectively. OS was similar to EFS for these groups. There were no local recurrences among patients with stage II AH. Four-year EFS and OS estimates for patients with bilateral AH (n = 29) were 43.8% (95% CI, 24.2 to 61.8) and 55.2% (95% CI, 34.8 to 71.7), respectively. CONCLUSION: The prognosis for patients with stage I AH is worse than that for patients with stage I FH. Novel treatment strategies are needed to improve outcomes for patients with AH, especially those with stage III to V disease. (05/2006)
• Breslow NE, Beckwith JB, Haase GM, Kalapurakal JA, Ritchey ML, Shamberger RC, Thomas PR, D'Angio GJ, Green DM. "Radiation therapy for favorable histology Wilms tumor: prevention of flank recurrence did not improve survival on National Wilms Tumor Studies 3 and 4.." Int J Radiat Oncol Biol Phys 65.1 (2006): 203-209. PURPOSE: To determine whether radiation therapy (RT) of patients with Wilms tumor of favorable histology prevented flank recurrence and thereby improved the survival outcomes. METHODS AND MATERIALS: Recurrence and mortality risks were compared among groups of patients with Stage I-IV/favorable histology Wilms tumor enrolled in the third (n = 1,640) and fourth (n = 2,066) National Wilms Tumor Study Group studies. RESULTS: Proportions of patients with flank recurrence were 0 of 513 = 0.0% for 20 Gy, 12 of 805 = 1.5% for 10 Gy, and 44 of 2,388 = 1.8% for no flank RT (p trend = 0.001 adjusted for stage and doxorubicin); for intra-abdominal (including flank) recurrence they were 5 of 513 = 1.0%, 30 of 805 = 3.7%, and 58 of 2,388 = 2.4%, respectively (p trend = 0.02 adjusted). Survival percentages at 8 years after intra-abdominal recurrence were 0 of 5 = 0% for 20 Gy, 10 of 30 = 33% for 10 Gy, and 34 of 58 = 56% for no RT (p trend = 0.0001). NWTS-4 discontinued use of 20 Gy RT, and the 8-year flank recurrence risk increased to 2.1% from 1.0% on NWTS-3 (p = 0.013). However, event-free survival was unaltered (88% vs. 86%, p = 0.39), and overall survival was better (93.8% vs. 90.8%, p = 0.036) on NWTS-4. CONCLUSIONS: Partly because of lower postrecurrence mortality among nonirradiated patients, prevention of flank recurrence by RT did not improve survival. It is important to evaluate entire treatment policies with regard to long-term outcomes. (05/2006)
• Grundy PE, Breslow NE, Li S, Perlman E, Beckwith JB, Ritchey ML, Shamberger RC, Haase GM, D'Angio GJ, Donaldson M, Coppes MJ, Malogolowkin M, Shearer P, Thomas PR, Macklis R, Tomlinson G, Huff V, Green DM. "Loss of heterozygosity for chromosomes 1p and 16q is an adverse prognostic factor in favorable-histology Wilms tumor: a report from the National Wilms Tumor Study Group.." J Clin Oncol 23.29 (2005): 7312-7321. PURPOSE: To determine if tumor-specific loss of heterozygosity (LOH) for chromosomes 1p or 16q is associated with a poorer prognosis for children with favorable-histology (FH) Wilms tumor entered on the fifth National Wilms Tumor Study (NWTS-5). PATIENTS AND METHODS: Between August 1995 and June 2002, 2,021 previously untreated children with FH or anaplastic Wilms tumor, clear-cell sarcoma of the kidney (CCSK) or malignant rhabdoid tumor of the kidney (RTK), were treated with stage- and histology-specific therapy. Their tumors were assayed for LOH for polymorphic DNA markers on chromosomes 1p and 16q.ResultsLOH for 1p or 16q was rarely observed in CCSK (n = 90) or RTK (n = 22). The relative risk (RR) of relapse for patients with FH stage I to IV tumors with LOH, stratified by stage, was 1.56 for LOH 1p (P = .01) and 1.49 for LOH 16q (P = .01), whereas the RR of death was 1.84 (P = .03) and 1.44 (P = .15), respectively. When the effects of LOH for both regions were considered jointly among patients with stage I to II FH disease, the risks of relapse and death were increased for LOH 1p only (RR = 2.2, P = .02 for relapse; RR = 4.0, P = .02 for death), for LOH 16q only (RR = 1.9, P = .01 and RR = 1.4, P = .60) and for LOH for both regions (RR = 2.9, P = .001 and RR = 4.3, P = .01) in comparison with patients with LOH at neither locus. The risks of relapse and death for patients with stage III to IV FH tumors were increased only with LOH for both regions (RR = 2.4, P = .01 and RR = 2.7, P = .04). CONCLUSION: Tumor-specific LOH for both chromosomes 1p and 16q identifies a subset of FH Wilms tumor patients who have a significantly increased risk of relapse and death. LOH for these chromosomal regions can now be used as an independent prognostic factor together with disease stage to target intensity of treatment to risk of treatment failure. (08/2005)

Scholarly Journals--Accepted

•  Beckwith JB: Antiquarian teratology books available in reprint form. Birth Defects Research A, (in press, scheduled publication in August 2009)   (08/2008 - 08/2009)
•  Beckwith JB. Smith’s recognizable patterns of human deformation (book review). Arch Pediatr Adolesc Med 162: (Accepted May, 1008).   (06/2007 - 06/2008)

Books and Chapters

•  Textbook Chapter: Weksberg R, Shuman C, Beckwith JB: Beckwith-Wiedemann Syndrome. In: Cassidy S (Ed.): Management of Genetic Syndromes (In Press).   (08/2008 - 08/2009)
•  Textbook Chapter: Argani P, Beckwith, JB: Renal Neoplasms of Childhood. in: Diagnostic Surgical Pathology (6th edition), Lippincott Williams & Wilkins, New York, (2009).     (08/2008 - 08/2009)
•  Testbook Chapter: Argani P, Beckwith, JB: Renal Neoplasms of Childhood. in: Diagnostic Surgical Pathology (5th edition), Lippincott Williams & Wilkins, New York, (in press).     (06/2007 - 06/2008)
• Book: Beckwith JB. Foundations of Teratology: Books and Artifacts Illustrating the History and Morphology of Congenital Malformations. New York: Oxford University Press, In Press, submitted February 2008, anticipated publication 2009.   (06/2007 - 06/2008)