Association of sinus histiocytosis with massive lymphadenopathy and idiopathic hypereosinophilic syndrome

A patient was evaluated because of edema, pruritus and generalized painless lymphadenopathy. Laboratory tests showed marked eosinophilia without known etiology. CT scan of abdomen revealed multiple lymph nodes in retroperitoneal area. Lymph node biopsy was reported as sinus histiocytosis, bone marrow biopsy showed hypercellularity with marked infiltration of normal eosinophils. During his admission he developed Coombs positive hemolytic anaemia. Once he was stable, a laparotomy was performed and the patient died two days later because of septic shock. Autopsy revealed sinus histiocytosis with massive lymphadenopathy (SHML) with extranodal involvement of duodenum, spleen and prostate; septic liver and spleen, pyelonephritis, marked infiltration of eosinophils in lymph nodes, spleen, liver duodenum and lungs. To the best of our knowledge, this is the first case report of the association of SHML and Idiopathic Hypereosinophilic Syndrome (HES).     

Problemas clínicos en micología médica: problema número 46

A 49 year old-man, without known previous diseases, who suffered an acute lung infection, is presented. He was a smoker and belonged to a high socioeconomic class. He suddenly presented high fever, dry cough and legs’ pain. Thorax X-ray and CT scan showed two excavated pneumonic infiltrates in the lower part of the right lung. He was treated with antibacterial antibiotics without clinical response. A transparietal fine needle lung biopsy was performed and the histopathology and cytological studies revealed an epithelioid granuloma with capsulated yeasts. Cryptococcus neoformans antigenemia was positive 1/128, and brain MR, as well as CSF studies, allowed to rule out a central nervous system compromise. The patient was successfully treated with itraconazole by oral route at a daily dose of 400 mg during 6 months. Post-treatment thorax CT scan showed a thin walled lung cavity as a sequela.     

Paracoccidioidomycosis Induced by Immunosuppressive Drugs in a Patient with Rheumatoid Arthritis and Bone Sarcoma: Case Report and Review of the Literature

Paracoccidioidomycosis (PCM) is a systemic mycosis caused by Paracoccidioides brasiliensis, which is endemic in many regions of Latin America. We describe the case of a 60-year-old man from a region endemic for PCM who presented with a long history of left hip pain. He had been treated over the past 3 years with immunosuppressive drugs (methotrexate, leflunomide, and adalimumab) for rheumatoid arthritis (RA). A hip radiograph showed lytic bone lesions, and a chest radiograph showed an expansive excavated lesion in the left lung, suggestive of a lung cancer with bone metastases. A left hip joint biopsy was inconclusive, but histological analysis of a surgical lung biopsy specimen was consistent with P. brasiliensis infection. Treatment with intravenous amphotericin B (50 mg/day) and hydrocortisone (25 mg/day) was initiated. However, increasing hip pain resulted in the amputation of the left lower limb, and the analysis of the surgical specimen revealed a diagnosis of bone sarcoma. Postoperatively, the patient developed sepsis and died approximately 1 month later. To our knowledge, this is the first report of PCM in a patient with RA who had been treated with immunosuppressive drugs, in particular TNF-α blocking agents. The atypical presentation (left hip pain alone) emphasizes the importance of considering PCM in the differential diagnosis of patients with pulmonary lesions and osteolytic lesions who live in a region endemic for PCM. This case report also demonstrates that health professionals in these regions must pay close attention to patients receiving immunosuppressive drugs because of the possibility of reactivating quiescent P. brasiliensis lesions.     

Disseminated histoplasmosis diagnosed by percutaneous needle biopsy of a retroperitoneal lymph node. A case report.

A case of disseminated histoplasmosis diagnosed by percutaneous needle biopsy cytology is reported. The patient presented with fever and pancytopenia. Computed tomography (CT) revealed retroperitoneal lymphadenopathy. Cytology smears prepared from a CT-guided screw needle biopsy of one of the lymph nodes showed numerous histiocytes with intracytoplasmic yeast forms consistent with Histoplasma capsulatum. Fungal cultures prepared from additional needle biopsy material confirmed the diagnosis. This case illustrates the utility of needle biopsy in the evaluation of radiographically detected retroperitoneal lymphadenopathy and in the rapid diagnosis of infectious disease in certain clinical settings.     

Scintigraphie des parathyroïdes double isotope Tc-MIBI/I dans l’hyperparathyroïdisme primaire : comparaison entre un protocole d’acquisition planaire et une TEMP de soustraction couplée à la TDM

In patient with primary hyperparathyroidism, preoperative imaging first objective is to determine accurately and reliably position and anatomic localization of enlarged parathyroid gland in order to guide surgeon for gland removal and to permit minimally invasive surgery. Although subtraction planar scintigraphy is an efficient imaging to diagnose hyperfunctioning parathyroid, the lack of anatomical reference for this single imaging technique could be obviated by hybrid imaging. We compared, in 23 patients with primary hyperparathyroidism, a planar scan of neck and mediastinum associated with a cervical anterior pinhole scan to a subtraction (¹²³I/^99mTc-MIBI) SPECT scan of neck and mediastinum fused with CT. Surgical and histopathologic findings were used as the standard of truth. Subtraction SPECT/CT appeared to be more sensitive and specific than planar protocol to diagnose and to accurately localize pathologic parathyroid. Sensitivity was 76 and 56 %, specificity was 100 and 80 % for hybrid SPECT/CT and planar protocol respectively. SPECT protocol was typically more efficient to diagnose pathologic parathyroid derived from upper gland and for patient with concomitant nodular goiter.     

Impact diagnostique de l’imagerie hybride TEMP/TDM dans la prise en charge des neuroblastomes. Expérience du service de médecine nucléaire du CHU Mohammed VI de Marrakech

IntroductionThe neuroblastoma is a malignant pediatric tumor of the peripheral sympathetic nervous system. This is the pediatric solid extracranial tumor most common and accounts for approximately 8 to 10% of childhood cancers. The diagnosis is based on imaging showing a tumor developed at the expense of the sympathetic nervous system, increased urinary catecholamines, increased uptake of meta-iodo-benzylguanidine (MIBG) and histology who finds a malignant proliferation of small round cells. An assessment of the tumor mass and its extension are required to assess the prognosis and adapt the treatment. The MIBG scintigraphy is a non-invasive imaging technique that can evaluate with a single review the totality of the tumor extension. The single-photon emission computed tomography/computed tomography (SPECT/CT) improves the sensitivity of the examination, it allows an anatomical and functional study and improves the anatomical localization of scintigraphic uptake observed. The purpose of our work is to illustrate the contribution of the SPECT/CT in addition to the planar MIBG scintigraphy in the diagnosis and staging of neuroblastoma about four cases.Case reportClinical case 1: a 4-year old child, presented for 2 months abdominal pain. In the clinical examination, he presented a hard abdominal mass, painless and right paramedian. Abdominal ultrasound showed a right retroperitoneal mass with lymph nodes. The dosage of urinary catecholamines was increased. The ¹³¹I-MIBG scintigraphy showed an image for a right adrenal neuroblastoma measuring 8.6 × 4.5 cm. Surgical excision of the adrenal mass revealed in the anatomopathologic study a malignant tumoral proliferation with round cells compatible with a neuroblastoma. Clinical case 2: a 10-month old infant, presented since 15 days an exophtalmia with poor general status. The clinical examination showed a right abdominal mass and a bilateral periorbital ecchymosis. The radiography of the thorax showed a widening mediastinal. Abdominal echography showed a tissular mass of the right adrenal gland with retroperitoneal lymph nodes and an ascites. The dosage of urinary catecholamines was increased. The ¹³¹I-MIBG scintigraphy showed a right adrenal neuroblastoma measuring 6.4 × 2.7 cm with orbital bone metastases in favor of a Hutchinson syndrome. Clinical case 3: a 2-month old infant, followed since 1 month for bilateral adrenal neuroblastoma. The clinical examination showed an important abdominal distension with bluish nodules under skin. The abdominal echography and the abdomino-pelvic TDM showed two adrenal masses corresponding to a bilateral neuroblastoma with liver metastases. The ¹³¹I-MIBG scintigraphy showed two adrenal masses measuring respectively 6.5 × 3.4 cm and 8 × 6 cm, with liver and skin metastases in favor of 4S neuroblastoma with bilateral adrenal tumors. Clinical case 4: a 3-year old child, followed for left adrenal neuroblastoma with multiple bone metastases. The clinical examination showed a left abdominal mass with exophtalmia and right palpebral ecchymosis. The radiography of the thorax showed a widening of the mediastin with repression of the paravertebral right line. Abdominal echography showed a left retroperitoneal tissular mass measuring 9.2 × 5.2 cm. The abdomino-pelvic TDM showed a left adrenal tumor with lumbar vertebral bone metastases. A first ¹³¹I-MIBG scan showed a left neuroblastoma with multiple bone metastases in the right orbit, the right humerus, the occipital bone, the right scapula and spine (D10, D11, L1, L3, S1). The child was treated by 5 courses of chemotherapy (protocol HRNLB/10). The ¹³¹I-MIBG scan control showed a regression of neuroblastoma size (1.4 × 1.2 cm) with loss of bone metastases of the occipital bone, the right scapula and spine.DiscussionThe MIBG scintigraphy is a simple, non-invasive examination that has excellent sensitivity and specificity in detection of neuroblastoma and especially in invasion bone marrow and in evaluation of the therapeutic response. The hybrid SPECT/CT imaging improve the performance of the scintigraphy as well in sensibility, toward the deep localization, as in specificity for images poorly defined in planar imaging.     

A Case Study of a Patient with Gliosarcoma with an Extended Survival and Spinal Cord Metastases

Gliosarcoma is a rare brain tumor consisting of both glial and mesenchymal components. Metastatic gliosarcoma is rare; however, here we report a 31-year-old Chinese woman with cranial gliosarcoma metastatic to the liver, lymph nodes and the spinal cord. Initially, the patient presented with dizziness, headache and vomiting and after surgery and histological examination, was diagnosed with cranial gliosarcoma. The patient was treated with surgical resection followed by chemotherapy and radiotherapy. Three years after completing treatment, the patient again presented with similar symptoms with the addition of a seizure. Test revealed recurrence of the gliosarcoma, and the same treatment was prescribed. Three years after treatment completion, the patient again presented with dizziness and headache. Masses at the right temple and in the right side of the neck were found. Tumors were surgically removed from the brain, skull, scalp and neck, the latter three diagnosed as metastatic gliosarcomas. The patient received both chemotherapy and radiotherapy following resection. One month after treatment, bone scans revealed possible metastasis in the right skull, lumbar and left ileum, soft neck tissue, lungs, collarbone, humeri, vertebrae, liver and abdominal lymph nodes. No further therapy was recommended due to the poor condition of the patient. The patient died 5 months later.     

Diffuse malignant lymphoma type B with optic chiasm infiltration, visual disturbances, hypopituitarism, hyperprolactinaemia and diabetes insipidus. Case report and literature review

The case is reported of a 55-year-old man with diffuse malignant lymphoma type B associated with transient optic chiasm infiltration and visual disturbances but with persistent hypopituitarism, hyperprolactinaemia and diabetes insipidus. The patient was administered chemotherapy and radiotherapy. Repeated MR and CT scans showed optic chiasm infiltration, which disappeared in the course of the chemotherapy but then recurred, changed its appearance and finally disappeared again. In the meantime visual disturbances occurred and disappeared during the therapy. Hypopituitarism, diabetes insipidus and hyperprolactinaemia were diagnosed and replacement therapy was administered. Later on abdominal pain occurred, and a CT scan revealed bilateral kidney masses and enlarged retroperitoneal lymph nodes. These were diffuse malignant lymphoma with regional lymphonodulitis in histology. Finally, hydrothorax and hydroretroperitoneum were diagnosed. The patient died as a result of systemic complications of the disease. The length of survival time documented following the hypothalamochiasmatic infiltration and diagnosis of lymphoma makes the case an unusual one for patients with CNS lymphoma. Hormonal disturbances accompanying the suprasellar region infiltration are very important from the practical point of view.     

Un examen au 18F-FDG d’interprétation délicate : une histoire de diaphragme

We present a clinical case illustrating the difficulty of interpretation of hypermetabolic foci localisation in 18F-FDG-PET/CT exam performed without injection of iodinated contrast. In a patient with cystic fibrosis during infection, the focus appears to be pathologic in subdiaphragmatic and retroperitoneal region. After a careful reading, the focus represents in reality the pillars diaphragmatic muscle attachments. The presence of other uptake in intercostal muscles in this patient presenting with respiratory failure and especially the comparison with an enhanced CT scan demonstrated our assumption.     

Phaeohyphomycosis Caused by Chaetomium Globosum in an Allogeneic Bone Marrow Transplant Recipient

Bone marrow transplant recipients are highly susceptible to opportunistic fungal infections. This is the report, of the first case of a Chaetomium systemic infection described in Brazil. A 34 year-old patient with chronic myeloid leukemia underwent an allogeneic sibling matched bone marrow transplant. Seven months later, he developed systemic infection with enlargement of the axillary and cervical lymph nodes. Culture of the aspirates from both lymph nodes yielded Chaetomium globosum. The infection was successfully treated with amphotericin B. The increasing population of immunosupressed patients requires a careful microbiologic investigation for uncommon fungal infections.     

Pure choriocarcinoma of the testis presenting with jaundice: a case report and review of the literature

ABSTRACT: INTRODUCTION: Testicular cancer is the most common malignancy in men 15- to 35-years-old. The North American standard classification divides testicular cancers into germ cell tumors and non-germ cell tumors. The lymphatic spread of germ cell tumors usually involves the retroperitoneal lymph nodes. However, this spread to the retroperitoneum rarely involves the hepatic hilum. We describe an unusual case of metastatic choriocarcinoma of the testis that was clinically mimicked by a cholestatic jaundice. This is an unusual presentation of testicular cancer and, to the best of our knowledge, the first report of this kind in the literature. CASE PRESENTATION: A 28-year-old Moroccan man presented with a four-week history of progressive obstructive jaundice, and weight loss to our emergency department. Abdominal ultrasound showed a dilatation of the biliary ducts due to pathologically enlarged lymph nodes of the hepatic hilum. A complete clinical and radiologic assessment to discover the primary tumor was negative except for pulmonary metastasis. In the laboratory findings at admission there were signs of cholestasis with an abnormal increase in the rate of testicular tumor markers (serum beta-human chorionic gonadotropin level was 11,000IU/ml), which subsequently led to the suspicion of a testicular tumor. Further evaluation included testicular palpation and ultrasound which revealed a testicular nodule. The patient underwent an inguinal orchidectomy of the right testis and histopathological examination confirmed a pure choriocarcinoma. The prognosis was poor due to lymph node involvement at the hepatic hilum. He died one month later, despite general chemotherapy. CONCLUSIONS: The clinical presentation of the disease and the rarity of this entity are two remarkable characteristics described in this case report which are rarely reported in literature.     

Apport de la TEMP/TDM pour la détection du ganglion sentinelle dans les cancers du col utérin et de l’endomètre

The sentinel lymph node procedure is still under evaluation for the management of cervical and endometrial carcinomas. The aim of our study was to determine the diagnostic accuracy of single-photon emission computed tomography/computed tomography (SPECT/CT) for preoperative sentinel lymph node mapping in uterine cancers. Sixty-eight patients with cervical (n = 42) or endometrial carcinoma (n = 26) underwent preoperative lymphoscintigraphy for sentinel node mapping. Sentinel node detection rate with conventional planar imaging was similar to that of SPECT/CT (87.1 versus 91.8 %) in the whole cohort. However, SPECT/CT detected a higher number of sentinel nodes in more than one third of patients, affected by either cervical or endometrial carcinoma. The rate of non or insufficiently contributive procedures (lack of uptake or unilateral uptake) in endometrial carcinomas was 47 % with conventional planar imaging, and 30 % with SPECT/CT. Sensitivity of both procedures for the detection of metastatic nodes was 81.8 %, compared to 100 % for the intraoperative combined detection (gamma probe sonde and blue dye). The impact of SPECT/CT for the sentinel lymph node detection in cervical and endometrial carcinomas needs further evaluation. Nevertheless, SPECT/CT may provide additional information when conventional planar imaging detects only unilateral uptake, may improve identification of atypical localizations, and facilitate surgical approach.     

Interferon-gamma and interleukin-4 single nucleotide gene polymorphisms in Paracoccidioidomycosis

The gene polymorphisms interferon-gamma (IFN-γ) +874 T/A and interleukin (IL)-4 −590 C/T have been associated with the altered production of cytokines. Therefore, they might be indicative of the occurrence of Paracoccidioidomycosis (PCM) caused by Paracoccidioides brasiliensis. The analysis of single nucleotide polymorphism (SNP) at position +874 IFN-γ showed an increase occurrence of A/T genotype in both PCM patients and healthy individuals as control (HIC) (56% and 45%, respectively), while the allelic distribution showed 82% of A allele in the patients and 80% in the controls. The SNP of −590 IL-4 showed that C/T genotype was significantly (p < 0.05) more prevalent (39%) in PCM group compared to the HIC group (19%), while IL-4 C/C genotype was significantly less frequent (59%) in the patient group compared to the control group (81%). Otherwise, 41% of PCM patients and 19% of HIC individuals carried the IL-4 T allele. Stimulation of peripheral blood mononuclear cells (PBMC) from PCM patients with cell extract antigenic preparations (PbAg) as well as secreted and surface antigens (MEXO) of P. brasiliensis evidenced that there is no difference in the IFN-γ production related to A and T alleles between PCM and HIC individuals. However, with IL-4 production, PCM patients classified as C phenotype showed two times more IL-4 production than PCM patients classified as T phenotype and HIC controls. In conclusion, our results suggest that functional genetic variants in the IL-4 promoter could influence the production of IL-4 in PCM.     

Incidental detection of a well-differentiated rectal adenocarcinoma by [18F]-Fluorocholine PET/CT in a patient with biochemical relapse of a prostate cancer

We report the case of a 70-year-old man who underwent an 18[F]-Fluorocholine ([18F]-FCH) positron emission tomography combined with computed tomography (PET/CT) after biochemical recurrence of prostate cancer previously treated by prostatectomy in 2012 (Gleason score 5 [2 + 3], PSA value before prostatectomy 19 ng/mL). Early acquisitions centered on the pelvis following the injection of 258 MBq of [18F]-FCH showed a rectal focused and intense uptake. The imaging performed 60 minutes after injection from the base of the skull to the upper third of the femur confirmed the presence of this focal uptake related on CT slices to a thickening of the rectal wall. Biopsies of the corresponding lesion allowed the diagnosis of well-differentiated rectal adenocarcinoma. The patient was directed to surgical resection, which was effective in September 2014. Pathology analysis was in favor of a well-differentiated rectal adenocarcinoma invaded the totality of the rectal wall. Fifteen lymph nodes were dissected and were benign. The tumor was classified as T3N0 (UICC 2010 classification). A contrast enhancement CT scan did not show any lesion compatible with rectal cancer metastasis. More and more incidental findings will occur in clinical PET/CT practice. If there are suspicious lesions of a different etiology than that announced in the request, nuclear medicine physicians must be aware of such issues in order to help clinicians to pursue further investigations like endoscopic biopsy for colorectal uptake. Finally, further studies must be performed to confirm the usefulness of [18F]-FCH PET/CT in colorectal cancer compared to [18F]-FDG PET/CT.     

Investigating centering,scan length,and arm position impact on radiation dose across 4 countries from 4 continents during pandemic: Mitigating key radioprotection issues

PurposeOptimization of CT scan practices can help achieve and maintain optimal radiation protection. The aim was to assess centering, scan length, and positioning of patients undergoing chest CT for suspected or known COVID-19 pneumonia and to investigate their effect on associated radiation doses.MethodsWith respective approvals from institutional review boards, we compiled CT imaging and radiation dose data from four hospitals belonging to four countries (Brazil, Iran, Italy, and USA) on 400 adult patients who underwent chest CT for suspected or known COVID-19 pneumonia between April 2020 and August 2020. We recorded patient demographics and volume CT dose index (CTDI_vol) and dose length product (DLP). From thin-section CT images of each patient, we estimated the scan length and recorded the first and last vertebral bodies at the scan start and end locations. Patient mis-centering and arm position were recorded. Data were analyzed with analysis of variance (ANOVA).ResultsThe extent and frequency of patient mis-centering did not differ across the four CT facilities (>0.09). The frequency of patients scanned with arms by their side (11–40% relative to those with arms up) had greater mis-centering and higher CTDI_vol and DLP at 2/4 facilities (p = 0.027–0.05). Despite lack of variations in effective diameters (p = 0.14), there were significantly variations in scan lengths, CTDI_vol and DLP across the four facilities (p < 0.001).ConclusionsMis-centering, over-scanning, and arms by the side are frequent issues with use of chest CT in COVID-19 pneumonia and are associated with higher radiation doses.     

A maximum pseudo-likelihood approach for estimating species trees under the coalescent model

Background  

Several phylogenetic approaches have been developed to estimate species trees from collections of gene trees. However, maximum likelihood approaches for estimating species trees under the coalescent model are limited. Although the likelihood of a species tree under the multispecies coalescent model has already been derived by Rannala and Yang, it can be shown that the maximum likelihood estimate (MLE) of the species tree (topology, branch lengths, and population sizes) from gene trees under this formula does not exist. In this paper, we develop a pseudo-likelihood function of the species tree to obtain maximum pseudo-likelihood estimates (MPE) of species trees, with branch lengths of the species tree in coalescent units.     

Carcinoma and multiple lymphomas in one patient: establishing the diagnoses and analyzing risk factors

Multiple malignancies may occur in the same patient, and a few reports describe cases with multiple hematologic and non-hematologic neoplasms. We report the case of a patient who showed the sequential occurrence of four different lymphoid neoplasms together with a squamous cell carcinoma of the lung. A 62-year-old man with adenopathy was admitted to the hospital, and lymph node biopsy was positive for low-grade follicular lymphoma. He achieved a partial remission with chemotherapy. Two years later, a PET-CT scan showed a left hilar mass in the lung; biopsy showed a squamous cell carcinoma. Simultaneously, he was diagnosed with diffuse large B cell lymphoma in a neck lymph node; after chemo- and radiotherapy, he achieved a complete response. A restaging PET-CT scan 2 years later revealed a retroperitoneal nodule, and biopsy again showed a low-grade follicular lymphoma, while a biopsy of a cutaneous scalp lesion showed a CD30-positive peripheral T cell lymphoma. After some months, a liver biopsy and a right cervical lymph node biopsy showed a CD30-positive peripheral T cell lymphoma consistent with anaplastic lymphoma kinase-negative anaplastic large cell lymphoma. Flow cytometry and cytogenetic and molecular genetic analysis performed at diagnosis and during the patient’s follow-up confirmed the presence of two clonally distinct B cell lymphomas, while the two T cell neoplasms were confirmed to be clonally related. We discuss the relationship between multiple neoplasms occurring in the same patient and the various possible risk factors involved in their development.     

TEP/TDM dans le diagnostic et la stadification des lymphomes

Histological subtypes of lymphomas are important because FDG uptake is much greater in aggressive than in indolent lymphomas and this, results in lower sensitivity of PET for the staging of indolent lymphomas. Staging is especially useful when treatment is changed according to staging. Staging with imaging methods has traditionally been performed using a CT scanner and has been based on the detection of nodal enlargement, an increased number of small nodes and in the presence of extranodal masses. However, CT is limited by its poor sensitivity in the detection of extranodal sites of involvement, in the identification of tumour involvement of normal size lymph nodes and in the differentiation between malignant and inflammatory enlarged lymph nodes. The uptake of FDG detected with PET images reflects metabolic activity rather than the size of the tissue masses, localizing tumoral activity in enlarged and in normal size lymph nodes. In the literature review that compares PET with CT, PET usually indicates more lesions than CT would and PET improves sensitivity without losing specificity. However, the majority of studies report that PET, improves the staging in a relatively limited number of patients (10-20%) and may change treatment in less than 10% of patients. Diagnostic accuracy of PET may improve with the use of hybrid PET/CT systems that combine metabolic and morphological imaging, in the same scanner and without moving the patient. This is a promising technique that will overcome the limitations of both modalities and may enhance diagnostic accuracy in lymphoma patients. This hybrid equipment allows the use of PET/CT with contrast-enhanced full dose CT (a diagnostic CT) instead of carrying out PET and CT on different days.