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Lv HY, Liu MX, Hong WT, Li XW. Primary hepatic neuroendocrine tumor with a suspicious pulmonary nodule: A case report and literature review. World J Clin Oncol 2025; 16:101236. [PMID: 40130063 PMCID: PMC11866086 DOI: 10.5306/wjco.v16.i3.101236] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/08/2024] [Revised: 12/07/2024] [Accepted: 12/27/2024] [Indexed: 01/21/2025] Open
Abstract
BACKGROUND Primary hepatic neuroendocrine tumors (PHNETs) are extremely rare tumors originating from neuroendocrine cells. Due to lack of neuroendocrine symptoms and specific radiographic characteristics, PHNETs are challenging to differentiate from other liver tumors. CASE SUMMARY This case involved a 67-year-old male who was admitted with a discovered hepatic mass and a suspicious lung lesion. Primary hepatic carcinoma was initially speculated based on the characteristic magnetic resonance imaging findings. The patient underwent a laparoscopic right partial hepatectomy, and subsequent immunohistochemical examination revealed a HNET. To exclude other potential origins, a positron emission tomography-computed tomography scan and gastrointestinal endoscopy were performed, leading to a final diagnosis of PHNETs. Then we conducted a literature review using the PubMed database, identifying 99 articles and 317 cases related to PHNETs. The characteristics, diagnostic methods, and treatment of PHNETs have been described. Finally, we elaborate on the presumed origins, pathological grades, clinical features, diagnosed methods, and treatments associated with PHNETs. CONCLUSION The diagnosis of PHNETs was primarily an exclusionary process. A definitive diagnosis of PHNETs relied mainly on immunohistochemical markers (chromogranin A, synaptophysin, and cluster of differentiation 56) and exclusion of primary foci in other organs. Radical surgery was the preferred treatment for early-stage tumors.
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Affiliation(s)
- Hai-Yan Lv
- Department of Nursing, The Second Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310009, Zhejiang Province, China
| | - Mei-Xuan Liu
- Department of Burns and Wound Care Center, The Second Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310009, Zhejiang Province, China
| | - Wen-Ting Hong
- Department of Nursing Pathology, The Second Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310009, Zhejiang Province, China
| | - Xia-Wei Li
- Department of Surgery, The Second Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310009, Zhejiang Province, China
- Key Laboratory of Cancer Prevention and Intervention, China National Ministry of Education, Cancer Institute, The Second Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310009, Zhejiang Province, China
- Department of Cancer Center, Zhejiang University, Hangzhou 310000, Zhejiang Province, China
- Harvard T.H. Chan School of Public Health, Harvard University, Cambridge, MA 02138, United States
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Fernández-Ferreira R, Romero-López U, Robles-Aviña JA, Rivas-Mendoza UN, González-Camacho C, Valero-Gómez A, Barquet-Mata OA, Reyes-Gabiño A, Tovar-Figueroa KA, Ramírez-Villagrán V. Primary Hepatic Neuroendocrine Carcinoma with Metastasis to the Mesentery: A Case Report. Case Rep Oncol 2023; 16:681-697. [PMID: 37933308 PMCID: PMC10625823 DOI: 10.1159/000533199] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/08/2023] [Accepted: 07/10/2023] [Indexed: 11/08/2023] Open
Abstract
Primary hepatic neuroendocrine carcinomas (PHNECs) are extremely rare, with only about 90 cases having been reported in the English-language literature. Among all neuroendocrine neoplasms, primary hepatic neuroendocrine tumors (NETs) and neuroendocrine carcinomas (NECs) are extremely rare, accounting for 0.3% of NETs and 0.28-0.46% of malignant liver tumors. Additionally, primary hepatic NECs occur infrequently. The clinical diagnosis of primary hepatic NEC remains challenging because of its rarity and the lack of information about its characteristic appearance on images. Consequently, pathological examination through the performance of a preoperative liver tumor biopsy is essential for diagnosis. Due to the lack of availability of substantial high-quality data, there is no standard therapy for primary hepatic NEC. We present the first case of PHNEC metastasized to the mesentery reported in the English-language literature.
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Affiliation(s)
- Ricardo Fernández-Ferreira
- Oncology Medicine and Surgical Oncology Department. Central South High Specialty Hospital PEMEX, Mexico City, Mexico
- Oncology Medicine and Surgical Oncology Department. Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
| | - Ulises Romero-López
- Faculty of Medicine. University of Veracruz. Minatitlan Campus. Heroica Veracruz, Mexico
| | - Jorge Alberto Robles-Aviña
- Oncology Medicine and Surgical Oncology Department. Central South High Specialty Hospital PEMEX, Mexico City, Mexico
| | - Uriel Norberto Rivas-Mendoza
- Oncology Medicine and Surgical Oncology Department. Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
| | - Casandra González-Camacho
- Imagenology Department, Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
| | - Alfredo Valero-Gómez
- Patology Department, Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
| | - Omar Armando Barquet-Mata
- Oncology Medicine and Surgical Oncology Department. Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
| | - Almira Reyes-Gabiño
- Oncology Medicine and Surgical Oncology Department. Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
| | - Karen Analí Tovar-Figueroa
- Oncology Medicine and Surgical Oncology Department. Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
| | - Viridiana Ramírez-Villagrán
- Oncology Medicine and Surgical Oncology Department. Tlahuac General Hospital Dr. Matilde Petra Montoya Lafragua, Mexico City, Mexico
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Seki Y, Sakata H, Uekusa T, Momose H, Yoneyama S, Hidemura A, Tajima Y, Suzuki H, Ishimaru M. Primary hepatic neuroendocrine carcinoma diagnosed by needle biopsy: a case report. Surg Case Rep 2021; 7:236. [PMID: 34727269 PMCID: PMC8563892 DOI: 10.1186/s40792-021-01315-3] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/31/2021] [Accepted: 10/19/2021] [Indexed: 12/18/2022] Open
Abstract
Background Primary hepatic neuroendocrine carcinomas (NECs) are extremely rare. The rate of recurrence after resection is extremely high, and the prognosis is poor. It is debatable whether chemotherapy or surgical resection is the optimal initial treatment for primary hepatic NECs. Therefore, selecting an appropriate therapeutic approach for patients with primary hepatic NECs remains clinically challenging. We present a case of primary hepatic NEC in a patient who developed recurrence after undergoing surgical resection. Case presentation A 78-year-old man with bone metastases of prostate cancer was referred to our department because of a solitary 66-mm tumor in the left lateral segment of the liver, which was detected on annual follow-up by computed tomography after prostate resection. A biopsy and preoperative diagnostic workup identified the lesion as a primary hepatic neuroendocrine carcinoma; therefore, left lateral segmentectomy was performed. Immunohistochemically, the tumor was positive for chromogranin A, synaptophysin, and CD 56, and the Ki-67 index was 40%. This neuroendocrine carcinoma was classified as a large cell type. Adjuvant chemotherapy with carboplatin + etoposide was initially administered a month after surgery. However, lymph node recurrence occurred 4 months after surgery, and the patient died of systemic metastases 15 months after surgical resection. Conclusions Due to the lack of availability of abundant quantities of relevant, high-quality data, there is no standard therapy for primary hepatic NECs. Selecting the most appropriate treatment for patients depending on several factors, such as the stage and differentiation of a tumor and a patient’s performance status and clinical course, is consequently preferred. More cases need to be studied to establish the best treatment strategy for primary hepatic NEC.
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Affiliation(s)
- Yusuke Seki
- Department of Surgery, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan.
| | - Hiroki Sakata
- Department of Surgery, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan
| | - Toshimasa Uekusa
- Department of Pathology, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan
| | - Hirokazu Momose
- Department of Hepato-Biliary-Pancreatic Surgery, Kyorin University Hospital, 6-20-2 Shinkawa, Mitaka City, Tokyo, 181-8611, Japan
| | - Satomi Yoneyama
- Department of Surgery, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan
| | - Akio Hidemura
- Department of Surgery, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan
| | - Yusuke Tajima
- Department of Surgery, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan
| | - Hiroyuki Suzuki
- Department of Surgery, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan
| | - Masahiro Ishimaru
- Department of Surgery, Kanto Rosai Hospital, 1-1 Kizukisumiyoshicho, Nakahara-ku, Kanagawa, 211-8510, Japan
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4
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Wang HH, Liu ZC, Zhang G, Li LH, Li L, Meng QB, Wang PJ, Shen DQ, Dang XW. Clinical characteristics and outcome of primary hepatic neuroendocrine tumors after comprehensive therapy. World J Gastrointest Oncol 2020; 12:1031-1043. [PMID: 33005296 PMCID: PMC7510005 DOI: 10.4251/wjgo.v12.i9.1031] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/08/2020] [Revised: 05/13/2020] [Accepted: 08/16/2020] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Primary hepatic neuroendocrine tumors (PHNETs), a group of neuroendocrine neoplasms, are extremely rare. There are only few case reports about PHNETs in the literature. The lack of large samples and multicenter research results in poor diagnostic and therapeutic approaches.
AIM To discuss the clinical characteristics, diagnosis, and treatment of PHNETs and risk factors related to survival.
METHODS We retrospectively analyzed the clinical data, imaging features, immunohistochemistry data, and treatment efficacy of 40 patients who were pathologically diagnosed with PHNETs and admitted to The First Affiliated Hospital of Zhengzhou University from January 1, 2014 to November 15, 2019. Finally, survival analysis was performed to identify the risk factors for survival.
RESULTS The main symptoms and signs included intermittent abdominal pain (19 patients, 47.5%) and bloating (8 patients, 20.0%). The positive rates of tested tumor markers were recorded as follows: Carbohydrate antigen 19-9 (CA19-9) (6 patients, 15.0%), CA72-4 (3 patients, 7.5%), carcinoembryonic antigen (7 patients, 17.5%), and alpha-fetoprotein (6 patients, 15.0%). Immunohistochemical staining results showed positivity for Syn in 38 (97.4%) of 39 patients, for chromogranin A in 17 (65.4%) of 26 patients, for CD56 in 35 (94.6%) of 37 patients, for AE1/AE3 in 28 (87.5%) of 32 patients, and for Ki-67 in all 40 (100.0%) patients. The overall survival rate was significantly related to the tumor grade, AE1/AE3, and Ki-67. No significant correlation was found between other parameters (age, gender, tumor number, tumor size, metastasis, and treatment) and overall survival.
CONCLUSION Higher grade, negative AE1/AE3, and higher Ki-67 are associated with a worse survival rate. Kinds of treatment and other parameters have no significant influence on overall survival.
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Affiliation(s)
- Hao-Hao Wang
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Zhao-Chen Liu
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Gong Zhang
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Lu-Hao Li
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Lin Li
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Qing-Bo Meng
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Pei-Ju Wang
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Dong-Qi Shen
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
| | - Xiao-Wei Dang
- Department of Hepatopancreatobiliary Surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450000, Henan Province, China
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Pastrián LG, Ruz-Caracuel I, Gonzalez RS. Giant Primary Neuroendocrine Neoplasms of the Liver: Report of 2 Cases With Molecular Characterization. Int J Surg Pathol 2019; 27:893-899. [DOI: 10.1177/1066896919855764] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
Primary neuroendocrine neoplasms of the liver have occasionally been reported in the liver, though many reports do not convincingly exclude metastases. In this article, we report 2 “giant” hepatic neuroendocrine lesions without evidence of a primary elsewhere after clinical workup. One occurred in a 21-year-old male; the lesion was a large cell neuroendocrine carcinoma measuring 24 cm. The patient died of disease in 10 months. The other occurred in a 25-year-old patient, was 18 cm wide, and was diagnosed as a well-differentiated neuroendocrine tumor, World Health Organization grade 3. The patient died of disease after 30 months. Molecular testing demonstrated only the presence of TP53 mutations in common. These cases expand our knowledge of seemingly primary neuroendocrine neoplasms of the liver, in particular, giant cases measuring more than 8 cm. Guidelines for clinical workup and therapy for these lesions remain unclear, but future thorough workup of such cases is necessary for specific characterization.
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Nakatake R, Ishizaki M, Matui K, Yanagimoto H, Inoue K, Kaibori M, Kawaguchi Y, Kon M. Combination therapies for primary hepatic neuroendocrine carcinoma: a case report. Surg Case Rep 2017; 3:102. [PMID: 28895097 PMCID: PMC5593802 DOI: 10.1186/s40792-017-0378-z] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/18/2017] [Accepted: 09/05/2017] [Indexed: 02/07/2023] Open
Abstract
Background Primary hepatic neuroendocrine carcinomas are extremely rare. Because of the rarity of PHNEC, its clinical features and treatment outcomes are not well understood. A proper diagnosis and the correct therapeutic approach therefore remain clinically challenging. Case presentation A 67-year-old man was admitted to our department because of a liver tumor. Computed tomography revealed a single liver tumor 50 mm in diameter and located in the S3 region. Biopsy and imaging findings resulted in a diagnosis of primary hepatic neuroendocrine carcinoma. Left lateral segmentectomy was performed. Immunohistochemically, the tumor cells were positive for synaptophysin, chromogranin A, and CD56. Ki-67 was positive in > 90% of the tumor cells. The final diagnosis was primary hepatic neuroendocrine carcinoma. The patient suffered two episodes of lymph node recurrence. Nonetheless, the tumor was excised to prolong survival. Thus, after lymphadenectomy, he received adjuvant chemotherapy for 6 months. Two years after surgery, the patient remains alive and in good general condition. Conclusions In most cases, primary hepatic neuroendocrine carcinoma, while extremely rare, has a poor prognosis. At present, surgical resection is a priority for curative treatment, but in patients with recurrence, combined therapies are recommended.
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Affiliation(s)
- Richi Nakatake
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan.
| | - Morihiko Ishizaki
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Kosuke Matui
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Hiroaki Yanagimoto
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Kentaro Inoue
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Masaki Kaibori
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Yusai Kawaguchi
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Masanori Kon
- Department of Surgery, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
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7
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Nishino H, Hatano E, Seo S, Shibuya S, Anazawa T, Iida T, Masui T, Taura K, Haga H, Uemoto S. Histological features of mixed neuroendocrine carcinoma and hepatocellular carcinoma in the liver: a case report and literature review. Clin J Gastroenterol 2016; 9:272-9. [PMID: 27384317 DOI: 10.1007/s12328-016-0669-0] [Citation(s) in RCA: 27] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/29/2016] [Accepted: 06/22/2016] [Indexed: 12/14/2022]
Abstract
Primary hepatic neuroendocrine carcinoma (NEC) is rare, and its origin, clinical features, diagnosis and treatment have not been clarified. Primary mixed NEC and hepatocellular carcinoma (HCC) is even rarer and is divided into either combined type or collision type. We report a patient with the combined type of mixed NEC and HCC. A 72-year-old male was diagnosed with HCC and underwent hepatectomy because of the presence of two tumors in his liver. Histological examination demonstrated that one of the tumors had both NEC and HCC components. The transitional zone was noted in the immunohistological examination, and the tumor was determined to be the combined type of primary mixed NEC and HCC. This was the first description of an HCC component being found in an NEC-dominant area in a primary mixed NEC and HCC tumor. Three weeks after hepatectomy, an abdominal computed tomography scan showed extensive regional and para-aortic lymphadenopathy, and early relapse was suspected in the patient. The patient died 3 months after the operation despite treatment with combination chemotherapy. This report describes an unfavorable case and summarizes all the primary mixed NEC and HCC tumor cases that have been previously reported.
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MESH Headings
- Aged
- Carcinoma, Hepatocellular/diagnostic imaging
- Carcinoma, Hepatocellular/pathology
- Carcinoma, Hepatocellular/surgery
- Carcinoma, Neuroendocrine/diagnostic imaging
- Carcinoma, Neuroendocrine/pathology
- Carcinoma, Neuroendocrine/surgery
- Hepatectomy/methods
- Humans
- Liver Neoplasms/diagnostic imaging
- Liver Neoplasms/pathology
- Liver Neoplasms/surgery
- Magnetic Resonance Imaging
- Male
- Neoplasms, Multiple Primary/diagnostic imaging
- Neoplasms, Multiple Primary/pathology
- Neoplasms, Multiple Primary/surgery
- Positron Emission Tomography Computed Tomography
- Tomography, X-Ray Computed
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Affiliation(s)
- Hiroto Nishino
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Etsuro Hatano
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan.
| | - Satoru Seo
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Shinsuke Shibuya
- Department of Diagnostic Pathology, Kyoto University Hospital, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Takayuki Anazawa
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Taku Iida
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Toshihiko Masui
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Kojiro Taura
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Hironori Haga
- Department of Diagnostic Pathology, Kyoto University Hospital, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
| | - Shinji Uemoto
- Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan
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Rocca A, Calise F, Marino G, Montagnani S, Cinelli M, Amato B, Guerra G. Primary giant hepatic neuroendocrine carcinoma: A case report. Int J Surg 2014; 12 Suppl 1:S218-21. [DOI: 10.1016/j.ijsu.2014.05.056] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/23/2014] [Accepted: 05/03/2014] [Indexed: 02/07/2023]
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