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Molecular Characterization of Juxtaglomerular Cell Tumors: Evidence of Alterations in MAPK-RAS Pathway.

Juxtaglomerular cell tumor (JGCT) is a rare neoplasm, part of the family of mesenchymal tumors of the kidney. Although the pathophysiological and clinical correlates of JGCT are well known, as these tumors are an important cause of early-onset arterial hypertension refractory to medical treatment, their molecular background is unknown, with only few small studies investigating their karyotype. Herein we describe a multi-institutional cohort of JGCTs diagnosed by experienced genitourinary pathologists, evaluating clinical presentation and outcome, morphologic diversity, and, importantly, the molecular features. Ten JGCTs were collected from 9 institutions, studied by immunohistochemistry, and submitted to whole exome sequencing. Our findings highlight the morphologic heterogeneity of JGCT, which can mimic several kidney tumor entities. Three cases showed concerning histologic features, but the patient course was unremarkable, which suggests that morphologic evaluation alone cannot reliably predict the clinical behavior. Gain-of-function variants in RAS GTPases were detected in JGCTs, with no evidence of additional recurrent genomic alterations. In conclusion, we present the largest series of JGCT characterized by whole exome sequencing, highlighting the putative role of the MAPK-RAS pathway.

Humans↗

Analysis of apolipoprotein A-I, lecithin:cholesterol acyltransferase and glucocerebrosidase genes in hypoalphalipoproteinemia.

Hypoalphalipoproteinemia (HALP) is a dyslipidemia characterized by low HDL-cholesterol (HDL-C) levels with important genetic contribution. However, no common genetic mutations have been found to be associated with this disorder. We screened the promoter and coding sequence of apolipoprotein (apo) A-I and lecithin:cholesterol acyltransferase (LCAT) genes and the 5' apo C-III region by SSCP and heteroduplex analysis, and DNA sequencing in 66 unrelated subjects with recurrent low HDL-C levels. We also analyzed the N370S and L444P variants, in the glucocerebrosidase (GBA) gene by restriction fragment analysis. Three mutations in the apo A-I gene (L144R, W108R, g.1833C>T) and 3 mutations in the LCAT gene (S208T, I178T, IVS3-23C>A) were detected, in six heterozygous subjects. In addition, a novel polymorphic site in LCAT gene (g.4886C>T) has been identified. Allelic frequencies of polymorphisms g.(-636)C>A, g.(-625)G>A, g.(-620)T>del, g.(-479C>T and g.(-452)T>C, located upstream of the apo C-III gene, were in normal range, and no other mutation was found in this region. Two HALP subjects were found to carry the N370S mutation at GBA locus. In conclusion, 12% of HALP subjects were found to carry mutations in apo A-I, LCAT, or GBA genes, which could explain this phenotype. Our results confirm the molecular, genetic and phenotypic heterogeneity of HALP.

Adult↗

Targetoid hemosiderotic hemangioma- a dynamic vascular tumor: report of 3 cases with episodic and cyclic changes and comparison with solitary angiokeratomas.

BACKGROUND: Both targetoid hemosiderotic hemangiomas (THH) and solitary angiokeratomas (SAK) are acquired vascular malformations formed by superficial vascular ectasias possibly caused by trauma. OBJECTIVE: We compare the clinicopathologic findings of THHs with those of SAKs and report the clinicopathologic findings of 3 singular cases of THH affected by cyclic or episodic morphologic changes. METHODS: We performed a clinicopathologic study on 33 cases of THH and compared this group with 20 cases of SAK. On selected cases, histochemical and immunohistochemical analyses were evaluated. RESULTS: Overlap of all the clinical and pathologic features studied were identified for THH and SAK. Clinically, they both commonly exhibited a brown or black papule located over the lower extremities that mimicked a melanocytic lesion. Histologically, they both had ectatic papillary dermal vessels with overlying epidermal hyperplasia, and adjacent hemosiderin deposits, extravasated red blood cells, lymphocytic infiltrate, and lymphangiectases. Compared with SAKs, THHs were significantly larger (5.3 vs 3.2 mm), more often excised (elliptical excision) than shave or punch biopsied, and had deeper dermal vessel alterations, more frequent dissecting vascular spaces, and more extensive hemosiderin deposits (all P < .01). THHs presenting with episodic changes were significantly larger than those without (11 vs 4.4 mm, P =.001). CONCLUSION: THHs and SAKs differ in degree, not in type, of clinicopathologic characteristics. This finding suggests that THHs are larger variants of SAKs whose size is the cause of more extensive, prolonged, or recurrent vessel damage. The histologic findings of extravasated red blood cells, hemosiderin, telangiectases, lymphangiectases, and fibrosis implicate trauma in the cause of these acquired vascular malformations.

Adolescent↗

A comparative analysis of FISH, RT-PCR, PCR, and immunohistochemistry for the diagnosis of mantle cell lymphomas.

Mantle cell lymphoma (MCL) diagnosis first relies on morphology and phenotype that may overlap with other B-cell lymphomas. Therefore, the demonstration of t(11;14)(q13;q32), the cytogenetic hallmark of MCL, is considered of diagnostic value. By studying a series of 35 MCL with characteristic morphology and phenotype (CD5+, CD10-, CD20+, CD23-), we have evaluated the applicability and the sensitivity of interphase fluorescence in situ hybridization (FISH) for t(11;14) detection and other techniques: (1) polymerase chain reaction (PCR) for amplification of t(11;14) genomic breakpoint, (2) competitive RT-PCR for the detection of cyclin D1 transcripts overexpression, and (3) immunohistochemistry (IHC) for cyclin D1 protein detection. Tissues from different origins were analyzed: lymph nodes (n = 24), spleen (n = 3), digestive biopsy (n = 3), tonsils (n = 3), and skin (n = 2). Interphase FISH was performed either on touch preparations (n = 11) and frozen (n = 9) or paraffin sections (n = 15). FISH analysis detected t(11;14) in 34/35 cases (97%) and demonstrated a recurrent CCND1 amplification in t(11;14)+ nuclei of the three blastoid MCL variants of our series. Genomic PCR analysis, hampered by the scattering of 11q13 breakpoints, was positive in only 13/35 cases (37%). RT-PCR analysis was applicable on nonepithelial tissues (27/35) and showed cyclin D1 transcript overexpression in all tested cases (27/35). IHC for cyclin D1 protein was performed either on frozen (n = 12) or on paraffin sections (n = 23), and its sensitivity was higher on paraffin sections (91%) than on frozen sections (25%). A cyclin D1 protein immunoreactivity was observed in 24/35 cases (69%). Our study emphasizes on the use of FISH analysis for the direct detection of t(11;14) because its applicability and sensitivity largely exceeded those of other techniques. It may also provide some informations on secondary cytogenetic changes of potential clinical relevance.

Antigens, CD20↗

Thermal capsulorraphy of bilateral glenohumeral joints in a pediatric patient with Ehlers-Danlos syndrome.

Ehlers-Danlos syndrome (EDS) is a heterogeneous collection of inherited connective tissue disorders characterized by hypermobility of the joints and hyperextensibility and fragility of the skin. For many patients, the hypermobile joints become problematic. To date, the mainstay of surgical treatment for EDS-related joint laxity has been open surgical capsulorraphy, which, although usually effective, confers significant morbidity to the patient. We present the case of a 9-year-old girl diagnosed with a variant of EDS and severely disabled from multidirectional instability of her shoulders and recurrent dislocations of her hips. After 1 year of nonoperative treatment (physical therapy, bracing, and activity restriction) failed, we performed a sequential arthroscopic thermal capsulorraphy of both shoulders. At a 2-year follow-up, the patient has no instability in the left shoulder and only occasional subluxations of the contralateral shoulder. We believe that thermal capsulorraphy is a viable addition to the shoulder surgeon's armamentarium in treating multidirectional instability in children with EDS.

Casts, Surgical↗

Central venulitis in pediatric liver allografts.

Central venulitis (CV), a distinct histologic lesion described in adult liver transplants, can occur with acute portal tract rejection or in isolation (ICV). Possible etiologies include immunosuppressive drug toxicity, acute cellular rejection, viral hepatitis, ischemic injury, and recurrent disease. This study was designed to characterize ICV and to assess its potential etiology in pediatric liver recipients because this population generally does not develop recurrent disease or viral hepatitis. All posttransplantation liver biopsy specimens that were obtained from children who received liver allografts over a 4-year period were reviewed. ICV was identified in 12 of 127 posttransplantation biopsies and in 7 of 45 allograft recipients. Only 4 liver transplantations were performed for potentially recurrent diseases (primary sclerosing cholangitis). ICV first appeared in posttransplantation biopsy specimens significantly later than did portal rejection alone. The finding of CV was not significantly correlated with elevation of Tacrolimus levels, reason for transplantation, donor/recipient cytomegalovirus (CMV) status or blood type, cold ischemic times, or the incidence of outflow obstruction. The responses of CV to therapy were variable and, although the majority of cases resolved, several episodes persisted or recurred. In conclusion, ICV occurs in 16% of pediatric liver allograft recipients and does not appear to be related to recurrent disease, viral hepatitis, drug toxicity, or graft ischemia. CV may be a variant of acute rejection, but longer follow-up is required to determine the most adequate therapy for this entity.

Adolescent↗

Transthyretin Leu12Pro is associated with systemic, neuropathic and leptomeningeal amyloidosis.

We report a middle-aged woman with a novel transthyretin (TTR) variant, Leu12Pro. She had extensive amyloid deposition in the leptomeninges and liver as well as the involvement of the heart and peripheral nervous system which characterizes familial amyloid polyneuropathy caused by variant TTR. Clinical features attributed to her leptomeningeal amyloid included radiculopathy, central hypoventilation, recurrent subarachnoid haemorrhage, depression, seizures and periods of decreased consciousness. MRI showed a marked enhancement throughout her meninges and ependyma, and TTR amyloid deposition was confirmed by meningeal biopsy. The simultaneous presence of extensive visceral amyloid and clinically significant deposits affecting both the peripheral and central nervous system extends the spectrum of amyloid-related disease associated with TTR mutations. The unusual association of severe peripheral neuropathy with symptoms of leptomeningeal amyloid indicates that leptomeningeal amyloidosis should be considered part of the syndrome of TTR-related familial amyloid polyneuropathy.

Adult↗

Reevaluation of corneal dystrophies of Bowman's layer and the anterior stroma (Reis-Bücklers and Thiel-Behnke types): a light and electron microscopic study of eight corneas and a review of the literature.

To clarify whether Reis-Bücklers' and Thiel-Behnke's corneal dystrophies are one entity or whether two distinct corneal dystrophies of Bowman's layer and the superficial stroma (CDB) exist, 28 corneal specimens with clinically suspected diagnosis of CDB were examined by light and electron microscopy, and the literature was reviewed. Eight specimens came from patients with a honeycomb-shaped pattern of opacities at the level of Bowman's layer. Study of those eight specimens disclosed destruction of Bowman's layer, a subepithelial fibrocellular tissue with an undulant configuration, absence of the epithelial basement membrane in many areas and the presence of "curly" collagen fibers with a diameter of 9-15 nm. We concluded that two distinct autosomal dominant CDB exist and propose the designations CDB type I (synonyms: geographic or "true" Reis-Bücklers' dystrophy) and CDB type II (synonyms: honeycomb-shaped or Thiel-Behnke's dystrophy). The eight corneas of our series were characterized as CDB type II. CDB I is clinically characterized by confluent geographic opacities at the level of Bowman's layer, histopathologically by bandshaped granular Masson-positive subepithelial deposits, and ultrastructurally by the presence of "rod-shaped bodies" and may be a superficial variant of granular dystrophy. Visual loss is significantly greater in CDB I, and recurrences after keratoplasty or keratectomy seem to be earlier and more extensive in CDB I. Most cases reported thus far in the literature as "Reis-Bücklers' dystrophy" are CDB II (honeycomb-shaped or Thiel-Behnke's dystrophy).

Adult↗

Is aseptic loosening truly aseptic?

Surgeons who treat osteomyelitis or infected implants think that microorganisms can live on and around implanted biomaterials and necrotic bone without clinical manifestations of infection. Gristina and Costerton, in their seminal work, suggested that such bacteria persist within biofilms and that they are often overlooked when diagnosis is based on standard microbiologic culture techniques. Subsequent studies using specialized techniques including sonication to remove adherent bacteria and direct detection using various forms of microscopy have confirmed that bacteria are present in many culture-negative cases. This led to the suggestion that at least some cases of failed orthopaedic implants that were considered aseptic loosening based on the absence of clinical signs of infection and the failure to isolate bacteria may actually have an infectious etiology. In addition to biofilms, potentially important concepts that also may contribute to false-negative culture results include the failure to recognize small colony variants induced during growth in vivo and the presence of bacteria inside host cells including osteoblasts. Importantly, bacteria persisting as small colony variants within biofilms and/or inside osteoblasts also may be an explanation for the recurrent nature of musculoskeletal infection.

Bacterial Infections↗

Immunodepression and the course of infection of a chronic Trypanosoma brucei infection in mice.

The relationships between course of infection, antigenic variation, and immunodepression of antibody responses to heterologous antigens have been investigated in mice chronically infected with Trypanosoma brucei. T. brucei Brunel University Trypanosomiasis (BUT) 64 produces a fluctuating parasitaemia lasting about 80 days and ending fatally. It is demonstrated that recurring peaks of parasitaemia are associated with the appearance of new variant antigenic types. At 21 and 31 days of infection, IgG responses to the heterologous antigen, sheep red blood cells (SRBC), are absent and IgM responses are less than 5% of normal. When a single dose of cyclophosphamide (300 mg/Kg) was injected into mice on day 31 of infection, the parasitaemia rose sharply in an uncontrolled fashion and the treated mice died in about 10 days. Cyclophosphamide, given in this way, is known to ablate antibody production completely but temporarily. It is therefore concluded that even though infected mice make extremely poor antibody responses to heterologous antigens, they are still capable of producing sufficient antibody to control peaks of parasitaemia associated with the emergence of new variant antigenic types. The significance of these findings is discussed in relation to recurrent hypotheses of trypanosome-associated immunodepression.

Animals↗

Micropapillary variant of transitional cell carcinoma of the bladder.

A 77-year-old man visited the Kobe City General Hospital complaining of macroscopic hematuria. A computed tomography scan found a bladder tumor with left iliac and para-aortic lymph node metastasis. Two courses of cisplatin, cyclophosphamide and doxorubicin chemotherapy resulted in a minimal response. Radical cystectomy and a retroperitoneal lymph node dissection with bilateral ureterocutaneostomy reconstruction were then performed. A pathological examination revealed a micropapillary variant of transitional cell carcinoma (Grade 3, pT1pN2M1). The patient died of pelvic recurrence 7 months after the initiation of chemotherapy. Peritonitis carcinomatosa and lung metastases were observed at autopsy.

Aged↗

Expression of the Cell Adhesion Molecule CD44 in Human Lung Tumors and Cell Lines.

Background: The purpose of this study was to examine the expression of the cell adhesion molecule CD44 in normal lung, primary and metastatic lung tumors, and cell lines derived from primary lung carcinomas. Methods and Results: A total of 68 lung specimens including normal tissue and primary and metastatic tumors, as well as 28 cell lines cultured from primary lung tumors with high recurrence, were examined for CD44 expression by semiquantitative reverse transcription polymerase chain reaction. Variant exon expression was confirmed by Southern blotting and hybridization of particular samples. In tumor tissues, loss of CD44 variant expression correlated with increasing tumor stage; a smaller percentage of more aggressive and poorly differentiated tumors expressed CD44v. Tumors metastatic to the lung were negative for CD44 variant expression. In primary lung cell lines, as in tumor tissue, tumors of higher histologic grade were characterized by loss of CD44 variant expression. Conclusion: CD44 isoform expression in normal lung and tumor tissues and cell lines revealed an overall decrease in CD44 alternative splicing in lung neoplasms of increased malignancy.

Journal Article↗

The spectrum of beta-thalassemia mutations in the Oran region of Algeria.

In order to delineate the spectrum of beta-globin gene defects causing beta-thalassemia in the Oran region of Algeria, we have analyzed a representative sample of 31 beta-thalassemia patients. This led to the detection of 10 mutations. Four of them [nonsense codon 39 (C->T), IVS-I-110 (G->A), IVS-I-2 (T->C), and frameshift codon 6 (-A)] account for approximately 77% of the beta-thalassemia chromosomes. Three of these mutants are also widespread in Mediterranean populations, whereas the fourth, IVS-I-2 (T->C), appears typical of the Oranese population. The six other variants are less frequent. The possible origin of these mutated alleles, either by recurrent mutational event or by migration from other populations, is discussed.

Adult↗

Homozygosity for the protein S Heerlen allele is associated with type I PS deficiency in a thrombophilic pedigree with multiple risk factors.

The multifactorial character of thrombotic disease is shown in a Spanish pedigree in which the propositus, with recurrent deep vein thrombosis, inherited the factor V R/Q506 mutation, the prothrombin 20210G/A variant and type III Protein S deficiency. Among 14 relatives carrying one or two of these three risk factors, thrombosis is present in a heterozygote for R/Q506 and in another for 20210G/A, who also had slightly positive antiphospholipid antibodies. Type I PS deficiency was also found in a young asymptomatic woman. PROS1 analysis showed coexistence of type III and type I PS deficiency to be associated with heterozygosity and homozygosity, respectively, for the P460 or PS Heerlen allele of the S/P460 variant. Analysis of PS values in this and other pedigrees segregating this variant revealed that not only free but also mean total PS levels are slightly but significantly lower in the SP460 heterozygotes than in the SS460 homozygotes. These findings strongly suggest a role of the P460 variant in the expression of the PS deficient phenotype.

Adult↗

[Pleuropulmonary blastoma in children: diagnosis and results of surgery and complex treatment].

This is a report on a retrospective study of experience had with complex treatment of pleuropulmonary blastoma in ten children aged 2-11 years, covering a 27-year period. In all patients diagnosis is made on the ground of clinical symptomatology, imaging methods and histological findings. All patients undergo surgery, as follows: tumor extirpation (n = 3), lobectomy (n = 2) and pneumonectomy (n = 5). Three deaths occur in the early postoperative period (30%). In the remainder postoperative chemotherapy is conducted. Histologically the solid variant (type 3 according to Dehner's classification) is predominant. There are three recurrences (42.8%) among the cases with simple tumor extirpation (2) and lobectomy (1), followed by successful pneumonectomy (two patients) and atypical pulmonary resection (one patient). At long-term follow-up, five patients (50%) are still alive for periods ranging from 1 to 11 years postoperatively. Pleuropulmonary blastoma is a surgical rarity in children. The most important factors for long-term survivorship are both radical surgery, and adequate chemotherapy.

Antineoplastic Combined Chemotherapy Protocols↗

Somatic-only SDHD variant with tumor-specific loss of heterozygosity in metastatic carotid body tumor: a case report with review of literature.

Carotid body tumors (CBTs) are rare paragangliomas in which genetic predisposition, particularly pathogenic variants in succinate dehydrogenase (SDHx) genes, plays an important role in tumorigenesis. Previous genetic studies of CBTs have primarily focused on germline SDHx variants, whereas somatic alterations remain poorly characterized. Among SDHx genes, SDHB variants are known to be associated with a higher metastatic risk, while SDHD variants are generally linked to a lower metastatic rate. We report the case of a 41-year-old man who presented with a painless right-sided neck mass. Imaging studies demonstrated a hypervascular tumor located at the carotid bifurcation with circumferential encasement of the carotid artery. During surgery, the tumor was classified as a Shamblin type III CBT, and complete surgical resection with vascular reconstruction was performed. Histopathological examination confirmed paraganglioma with metastasis to a single cervical lymph node. Germline genetic testing did not reveal any pathogenic variants. However, comprehensive tumor genomic profiling identified a somatic SDHD c.304C>G (p.His102Asp) variant accompanied by tumor-specific loss of heterozygosity (LOH) and copy-number loss at the SDHD locus, findings compatible with biallelic SDHD inactivation. The patient remained free of recurrence during follow-up. To our knowledge, this represents the first reported case of metastatic CBT harboring a somatic-only SDHD variant with tumor-specific LOH. This case suggests that reliance on germline testing alone may underestimate the molecular drivers of CBT and highlights the potential clinical value of tumor-based genomic profiling for risk stratification and prognostic assessment.

Carotid body tumor↗

[Venography of postoperative recurrence of symptomatic varicocele in males].

INTRODUCTION: We report the results of our experience with venography in patients with postoperative recurrent varicocele. The study was carried out to detect the causes of this condition. MATERIAL AND METHODS: Forty-four patients with postoperative recurrent varicocele, examined in our department from June, 1993, to June, 1996, were submitted to selective spermatic venography after clinical examination and color Doppler sonography. Thirty-six patients had been treated with high surgical ligation of the spermatic vein and 8 with inguinal ligation. Thirty-two patients were treated percutaneously, after diagnostic angiography, with coils and/or sclerotizing agents. RESULTS: In our study, the persistence of patent collateral veins, missed at surgical ligation, was the main cause of recurrence (68%): this was due either to a double or triple spermatic vein, mostly in the pelvic tract (50%), venous bridges crossing the surgical ligation (11%), or to retroperitoneal anastomoses (7%). A smaller group of patients showed ineffective ligation of the vein (27%); in the remaining 5% of cases incompetence of the extrafunicular plexus was detected. DISCUSSION AND CONCLUSIONS: Our experience, supported by a literature review, demonstrates that the anatomic variants, not detected preoperatively because phlebography had not been performed, were the most frequent causes of recurrence. Spermatic venography is the most accurate imaging modality for vascular mapping in postoperative recurrences; it often allows to treat the patients simultaneously by a percutaneous approach.

Adult↗