Editorial
When Encountering a Rare Vascular Disease, Report and Share your Experience: Six Reasons to Publish a Case Report in VSI
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Vasc Specialist Int (2023) 39:9
Published online March 31, 2023 https://doi.org/10.5758/vsi.233911
Copyright © The Korean Society for Vascular Surgery.
“You learn more from failure than from success. Don’t let it stop you. Failure builds character.”
Approximately 20 years ago, when I started my career as a vascular surgeon after fellowship training, I received an emergency call for a 52-year-old female with flu-like symptoms, fever, and painful swelling of her left neck and arm for 8 days. She had no history of trauma or central catheterization. Contrast-enhanced computed tomography revealed extensive acute thrombosis in the left internal jugular and subclavian veins. I had never seen such extensive venous thrombosis before. No one around me knew about this disease. I searched vascular textbooks; however, none were helpful. I proceeded to search the PubMed database using the search terms “thrombosis subclavian vein internal jugular vein.” It revealed more than 200 articles, the abstracts of which I read, concentrating on and searching for answers regarding diagnosis and treatment. Finally, I found an article dealing with Lemierre syndrome that exactly matched the clinical presentation of my patient. It took about 2 hours, and I left work with a happy heart after ordering antibiotics and anticoagulants. The following day, I attempted a more aggressive treatment approach via aspiration thrombectomy to obtain samples of infected thrombi for microbiological culture and to decrease the thrombus burden for earlier recanalization. However, the thrombi were thick and adhered to the vessel wall due to inflammation, and the retrieved thrombi revealed no microorganisms. After 6 weeks of antibiotics and 3 months of anticoagulant therapy, the veins completely recanalized, and her symptoms disappeared. Later, I realized that thrombolysis could be dangerous, causing multiple septic emboli, pneumonia, or systemic abscesses. After experiencing this rare case, I published a case report to share my knowledge [1]. This case report has been cited by at least 6 doctors and has helped more patients.
Herein, I would like to summarize why case reports of rare vascular diseases are needed, and how to write a good case report.
1) Report and share your experience to establish evidence-based practices
When a vascular case with a rare clinical presentation, unique imaging, novel treatment modality, or unexpected complications is encountered, it can be reported to a peer-reviewed journal such as Vasc Specialist Int (VSI). The current principles and ethics of modern medical practice are founded on evidence-based medicine, which means using the best available scientific evidence to inform clinical decision-making and improve patient outcomes. In the evidence pyramid, basic and fundamental evidence begins with case reports and case series. When encountering a rare vascular disease in practice, please report and share your experiences, especially in the VSI journal. Researchers can draw meaningful conclusions about rare diseases when enough cases are gathered. In addition, I recommend describing the case in detail, which may help in future collective analyses of the disease.
2) Be courageous to report a nightmare case
A famous quote by poet Alexander Pope states that “To err is human; to forgive, divine.” Sometimes, we learn more from failed cases than from successful ones. Complicated cases frequently provide critical lessons that were not expected preoperatively. Mistakes can be made based on human nature; continued medical education, training, and active surveillance of perioperative outcomes can help prevent or reduce mistakes.
Aleksic et al. [2] from Germany reported successful reconstruction of a stripped superficial femoral vein. The femoral vein was inadvertently stripped during varicose vein surgery, causing massive bleeding and shock. The stripped vein was rinsed with betadine and reimplanted, which remained patent for 11 months. van Nieuwenhuizen et al. [3] from the Netherlands reported a fatal case of small bowel perforation due to tunneling of a femorofemoral bypass. Their candid report revealed their mistakes in order to raise awareness of this rare fatal complication for educational purposes. Chung et al. [4] also reported a similar case for the same purpose. Reports of negative outcomes or complications are rare. Due to the reluctance of researchers to submit poor outcomes, resulting in publication bias, the potential harm of a procedure is usually underestimated or the treatment effects are overestimated, which can lead to flawed clinical guidelines or recommendations. To minimize the effect of publication bias, researchers should be encouraged to publish all research findings regardless of their results.
Oh et al. [5] reported 2 cases of meso-Rex shunt in children with extrahepatic portal vein occlusion. One case was successful, and the other failed; the reason for the different outcomes was obscure. The details of the 2 cases were reported for future analysis.
3) Publish or perish: make a record of historic novel treatments
In 2020, Ivancev and Vogelzang published an excellent editorial entitled “A 35 year history of stent grafting, and how EVAR conquered the world” [6]. However, their description of the world’s first fenestrated endovascular aneurysm repair (FEVAR) of the renal artery was not true. Therefore, I wrote an editorial in VSI [7], and a letter to the editor in the European Journal of Vascular and Endovascular Surgery [8]. The authors apologized for their omission. This correction was accepted because of a previous case report of 2 cases of the world’s first FEVAR performed in Seoul, Korea [9]. An old Korean proverb says that “Tigers leave their skins when they die, but humans leave their names.” Publish your scientific work or perish.
4) Report and remind readers of an unusual presentation of an exceptional case
In 2011, Kim et al. [10] reported an exceptional case of a wooden toothpick retrieved from an inferior vena cava (IVC) thrombus in a Caucasian woman with fever and abdominal pain. After successful endovascular removal and recovery without complications, the question remained as to how the toothpick traveled to the IVC. Speculations regarding the mysterious route of the toothpick into the IVC were investigated and discussed. A possible theory was that after ingesting a toothpick in street food, possibly under drug or alcohol intoxication, it penetrated the third portion of the duodenum and the nearby IVC, resulting in caval thrombophlebitis.
Kim et al. [11] reported a case of intravascular fasciitis in the femoral vein, mimicking a sarcoma with a happy ending. Park et al. [12] reported an intravascular epithelioid angiosarcoma in the abdominal aorta that mimicked an infected aneurysm. These cases are extremely rare to encounter, almost once in a lifetime, and reports from many researchers are important to define the clinical presentation and natural history of the disease.
5) Think rational, behave bravely: record innovative ideas and treatment of a rare vascular disease
Lee et al. [13] reported the first case of endovascular treatment for a celiomesenteric trunk (CMT) aneurysm. To commemorate the success of the first case of endovascular treatment of a CMT aneurysm, we reported the case using a cartoon of a giraffe, hoping for a good long-term prognosis (Fig. 1). After a comprehensive review of the literature, I created a table of 22 reported cases of CMT aneurysms for future researchers. This kind of work benefits not only surgeons and researchers encountering CMT aneurysms but also their patients.
-
Figure 1.Celiomesenteric trunk (CMT) aneurysm was treated by endovascular intervention, which appears like a giraffe’s head. Adapted from the article of Lee et al. (Vasc Specialist Int 2018;34:44-47) [13].
6) Make a collection of unique diseases in your country
In 2015, Choi et al. [14] reported 2 rare cases of IVC or aortic injury caused by acupuncture, a popular practice in Korea for treating back pain. Vascular surgeons should be aware that acupuncture can cause serious damage to the IVC or aorta, resulting in infection or chyle leakage. Case reports or case series comprise the most basic section of the evidence pyramid. Reporting rare vascular diseases is important for future patients and doctors.
VSI is a peer-reviewed, open-access international journal on vascular surgery, and any case reports, original articles, or review articles are welcome without any publication charges. In summary, there is a survival guide when a nobody-knows-disease is encountered:
Be courageous and remain positive.
Consult with experts and use available internet search engines or artificial intelligence-based platforms.
Find your rationale in treating the disease, and follow the Hippocratic Oath, “Do no harm.”
Report and share your experience for future patients and doctors.
CONFLICTS OF INTEREST
The author has nothing to disclose.
References
- Min SK, Park YH, Cho YK, Park JW, Koh YH, Seo TS. Lemierre's syndrome: unusual cause of internal jugular vein thrombosis--a case report. Angiology 2005;56:483-487.
- Aleksic I, Busch T, Sîrbu H, Tirilomis T, Bensch M, Dalichau H. Successful reconstruction of stripped superficial femoral vein. J Vasc Surg 2001;33:1111-1113.
- van Nieuwenhuizen RC, Kox C, Legemate DA. Small bowel perforation due to tunnelling of a femorofemoral crossover bypass. EJVES Extra 2007;13:21-22.
- Chung CTY, Min S, Min SK. Do not tunnel the small bowel during the tunneling of a femoro-femoral bypass. Vasc Specialist Int 2021; https://doi.org/10.5758/vsi.210036.
- Oh MY, Kim HK, Han A, Yi NJ, Min SK. Standard or variant Meso-Rex shunts for children with variceal bleeding due to extrahepatic portal vein obstruction: a report of two cases. Vasc Specialist Int 2020;36:186-192.
- Ivancev K, Vogelzang R. A 35 year history of stent grafting, and how EVAR conquered the world. Eur J Vasc Endovasc Surg 2020;59:685-694.
- Min SK, Chung JW. Forgotten stories of the first fenestrated endovascular aneurysm repair in human performed in 1995 and published in 1996. Vasc Specialist Int 2020;36:203-206.
- Min SK, Chung JW. Comment on "A 35 year history of stent grafting and how EVAR conquered the world". Eur J Vasc Endovasc Surg 2021;61:865.
- Park JH, Chung JW, Choo IW, Kim SJ, Lee JY, Han MC. Fenestrated stent-grafts for preserving visceral arterial branches in the treatment of abdominal aortic aneurysms: preliminary experience. J Vasc Interv Radiol 1996;7:819-823.
- Kim SY, Kim HC, Oh MD, Chung JW, Kim SJ, Min SK. Successful percutaneous thrombectomy of an infected vena-caval thrombus due to a toothpick. J Vasc Surg 2011;54:1498-1500.
- Kim HK, Han A, Ahn S, Min S, Ha J, Min SK. Intravascular fasciitis in the femoral vein with hypermetabolic signals mimicking a sarcoma: the role of preoperative imaging studies with review of literature. Vasc Specialist Int 2021;37:50-57.
- Park WK, Park KL, Cho YS, Han A, Ahn S, Min SK. Intravascular epithelioid angiosarcoma in the abdominal aorta mimicking an infected aneurysm. Vasc Specialist Int 2019;35:232-236.
- Lee SA, Jae HJ, Ahn S, Min SI, Ha J, Min SK. Endovascular treatment of a saccular aneurysm in the celiomesenteric trunk: a case report and review of literature. Vasc Specialist Int 2018;34:44-47.
- Choi C, Jung IM, Min SK, Han A, Ha J, Min SI. Inadvertent vascular injury of the aorta or vena cava caused by acupuncture. J Vasc Surg Cases 2015;1:13-15.
Related articles in VSI
Article
Editorial
Vasc Specialist Int (2023) 39:9
Published online March 31, 2023 https://doi.org/10.5758/vsi.233911
Copyright © The Korean Society for Vascular Surgery.
When Encountering a Rare Vascular Disease, Report and Share your Experience: Six Reasons to Publish a Case Report in VSI
Division of Vascular Surgery, Department of Surgery, Seoul National University College of Medicine, Seoul, Korea
Correspondence to:Seung-Kee Min, Division of Vascular Surgery, Seoul National University Hospital, 101 Daehak-ro, Jongno-gu, Seoul 03080, Korea
Tel: 82-2-2072-0297, Fax: 82-2-766-3975, E-mail: skminmd@snuh.org, https://orcid.org/0000-0002-1433-2562
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Body
“You learn more from failure than from success. Don’t let it stop you. Failure builds character.”
Approximately 20 years ago, when I started my career as a vascular surgeon after fellowship training, I received an emergency call for a 52-year-old female with flu-like symptoms, fever, and painful swelling of her left neck and arm for 8 days. She had no history of trauma or central catheterization. Contrast-enhanced computed tomography revealed extensive acute thrombosis in the left internal jugular and subclavian veins. I had never seen such extensive venous thrombosis before. No one around me knew about this disease. I searched vascular textbooks; however, none were helpful. I proceeded to search the PubMed database using the search terms “thrombosis subclavian vein internal jugular vein.” It revealed more than 200 articles, the abstracts of which I read, concentrating on and searching for answers regarding diagnosis and treatment. Finally, I found an article dealing with Lemierre syndrome that exactly matched the clinical presentation of my patient. It took about 2 hours, and I left work with a happy heart after ordering antibiotics and anticoagulants. The following day, I attempted a more aggressive treatment approach via aspiration thrombectomy to obtain samples of infected thrombi for microbiological culture and to decrease the thrombus burden for earlier recanalization. However, the thrombi were thick and adhered to the vessel wall due to inflammation, and the retrieved thrombi revealed no microorganisms. After 6 weeks of antibiotics and 3 months of anticoagulant therapy, the veins completely recanalized, and her symptoms disappeared. Later, I realized that thrombolysis could be dangerous, causing multiple septic emboli, pneumonia, or systemic abscesses. After experiencing this rare case, I published a case report to share my knowledge [1]. This case report has been cited by at least 6 doctors and has helped more patients.
Herein, I would like to summarize why case reports of rare vascular diseases are needed, and how to write a good case report.
1) Report and share your experience to establish evidence-based practices
When a vascular case with a rare clinical presentation, unique imaging, novel treatment modality, or unexpected complications is encountered, it can be reported to a peer-reviewed journal such as Vasc Specialist Int (VSI). The current principles and ethics of modern medical practice are founded on evidence-based medicine, which means using the best available scientific evidence to inform clinical decision-making and improve patient outcomes. In the evidence pyramid, basic and fundamental evidence begins with case reports and case series. When encountering a rare vascular disease in practice, please report and share your experiences, especially in the VSI journal. Researchers can draw meaningful conclusions about rare diseases when enough cases are gathered. In addition, I recommend describing the case in detail, which may help in future collective analyses of the disease.
2) Be courageous to report a nightmare case
A famous quote by poet Alexander Pope states that “To err is human; to forgive, divine.” Sometimes, we learn more from failed cases than from successful ones. Complicated cases frequently provide critical lessons that were not expected preoperatively. Mistakes can be made based on human nature; continued medical education, training, and active surveillance of perioperative outcomes can help prevent or reduce mistakes.
Aleksic et al. [2] from Germany reported successful reconstruction of a stripped superficial femoral vein. The femoral vein was inadvertently stripped during varicose vein surgery, causing massive bleeding and shock. The stripped vein was rinsed with betadine and reimplanted, which remained patent for 11 months. van Nieuwenhuizen et al. [3] from the Netherlands reported a fatal case of small bowel perforation due to tunneling of a femorofemoral bypass. Their candid report revealed their mistakes in order to raise awareness of this rare fatal complication for educational purposes. Chung et al. [4] also reported a similar case for the same purpose. Reports of negative outcomes or complications are rare. Due to the reluctance of researchers to submit poor outcomes, resulting in publication bias, the potential harm of a procedure is usually underestimated or the treatment effects are overestimated, which can lead to flawed clinical guidelines or recommendations. To minimize the effect of publication bias, researchers should be encouraged to publish all research findings regardless of their results.
Oh et al. [5] reported 2 cases of meso-Rex shunt in children with extrahepatic portal vein occlusion. One case was successful, and the other failed; the reason for the different outcomes was obscure. The details of the 2 cases were reported for future analysis.
3) Publish or perish: make a record of historic novel treatments
In 2020, Ivancev and Vogelzang published an excellent editorial entitled “A 35 year history of stent grafting, and how EVAR conquered the world” [6]. However, their description of the world’s first fenestrated endovascular aneurysm repair (FEVAR) of the renal artery was not true. Therefore, I wrote an editorial in VSI [7], and a letter to the editor in the European Journal of Vascular and Endovascular Surgery [8]. The authors apologized for their omission. This correction was accepted because of a previous case report of 2 cases of the world’s first FEVAR performed in Seoul, Korea [9]. An old Korean proverb says that “Tigers leave their skins when they die, but humans leave their names.” Publish your scientific work or perish.
4) Report and remind readers of an unusual presentation of an exceptional case
In 2011, Kim et al. [10] reported an exceptional case of a wooden toothpick retrieved from an inferior vena cava (IVC) thrombus in a Caucasian woman with fever and abdominal pain. After successful endovascular removal and recovery without complications, the question remained as to how the toothpick traveled to the IVC. Speculations regarding the mysterious route of the toothpick into the IVC were investigated and discussed. A possible theory was that after ingesting a toothpick in street food, possibly under drug or alcohol intoxication, it penetrated the third portion of the duodenum and the nearby IVC, resulting in caval thrombophlebitis.
Kim et al. [11] reported a case of intravascular fasciitis in the femoral vein, mimicking a sarcoma with a happy ending. Park et al. [12] reported an intravascular epithelioid angiosarcoma in the abdominal aorta that mimicked an infected aneurysm. These cases are extremely rare to encounter, almost once in a lifetime, and reports from many researchers are important to define the clinical presentation and natural history of the disease.
5) Think rational, behave bravely: record innovative ideas and treatment of a rare vascular disease
Lee et al. [13] reported the first case of endovascular treatment for a celiomesenteric trunk (CMT) aneurysm. To commemorate the success of the first case of endovascular treatment of a CMT aneurysm, we reported the case using a cartoon of a giraffe, hoping for a good long-term prognosis (Fig. 1). After a comprehensive review of the literature, I created a table of 22 reported cases of CMT aneurysms for future researchers. This kind of work benefits not only surgeons and researchers encountering CMT aneurysms but also their patients.
-
Figure 1. Celiomesenteric trunk (CMT) aneurysm was treated by endovascular intervention, which appears like a giraffe’s head. Adapted from the article of Lee et al. (Vasc Specialist Int 2018;34:44-47) [13].
6) Make a collection of unique diseases in your country
In 2015, Choi et al. [14] reported 2 rare cases of IVC or aortic injury caused by acupuncture, a popular practice in Korea for treating back pain. Vascular surgeons should be aware that acupuncture can cause serious damage to the IVC or aorta, resulting in infection or chyle leakage. Case reports or case series comprise the most basic section of the evidence pyramid. Reporting rare vascular diseases is important for future patients and doctors.
VSI is a peer-reviewed, open-access international journal on vascular surgery, and any case reports, original articles, or review articles are welcome without any publication charges. In summary, there is a survival guide when a nobody-knows-disease is encountered:
Be courageous and remain positive.
Consult with experts and use available internet search engines or artificial intelligence-based platforms.
Find your rationale in treating the disease, and follow the Hippocratic Oath, “Do no harm.”
Report and share your experience for future patients and doctors.
CONFLICTS OF INTEREST
The author has nothing to disclose.
Fig 1.
References
- Min SK, Park YH, Cho YK, Park JW, Koh YH, Seo TS. Lemierre's syndrome: unusual cause of internal jugular vein thrombosis--a case report. Angiology 2005;56:483-487.
- Aleksic I, Busch T, Sîrbu H, Tirilomis T, Bensch M, Dalichau H. Successful reconstruction of stripped superficial femoral vein. J Vasc Surg 2001;33:1111-1113.
- van Nieuwenhuizen RC, Kox C, Legemate DA. Small bowel perforation due to tunnelling of a femorofemoral crossover bypass. EJVES Extra 2007;13:21-22.
- Chung CTY, Min S, Min SK. Do not tunnel the small bowel during the tunneling of a femoro-femoral bypass. Vasc Specialist Int 2021; https://doi.org/10.5758/vsi.210036.
- Oh MY, Kim HK, Han A, Yi NJ, Min SK. Standard or variant Meso-Rex shunts for children with variceal bleeding due to extrahepatic portal vein obstruction: a report of two cases. Vasc Specialist Int 2020;36:186-192.
- Ivancev K, Vogelzang R. A 35 year history of stent grafting, and how EVAR conquered the world. Eur J Vasc Endovasc Surg 2020;59:685-694.
- Min SK, Chung JW. Forgotten stories of the first fenestrated endovascular aneurysm repair in human performed in 1995 and published in 1996. Vasc Specialist Int 2020;36:203-206.
- Min SK, Chung JW. Comment on "A 35 year history of stent grafting and how EVAR conquered the world". Eur J Vasc Endovasc Surg 2021;61:865.
- Park JH, Chung JW, Choo IW, Kim SJ, Lee JY, Han MC. Fenestrated stent-grafts for preserving visceral arterial branches in the treatment of abdominal aortic aneurysms: preliminary experience. J Vasc Interv Radiol 1996;7:819-823.
- Kim SY, Kim HC, Oh MD, Chung JW, Kim SJ, Min SK. Successful percutaneous thrombectomy of an infected vena-caval thrombus due to a toothpick. J Vasc Surg 2011;54:1498-1500.
- Kim HK, Han A, Ahn S, Min S, Ha J, Min SK. Intravascular fasciitis in the femoral vein with hypermetabolic signals mimicking a sarcoma: the role of preoperative imaging studies with review of literature. Vasc Specialist Int 2021;37:50-57.
- Park WK, Park KL, Cho YS, Han A, Ahn S, Min SK. Intravascular epithelioid angiosarcoma in the abdominal aorta mimicking an infected aneurysm. Vasc Specialist Int 2019;35:232-236.
- Lee SA, Jae HJ, Ahn S, Min SI, Ha J, Min SK. Endovascular treatment of a saccular aneurysm in the celiomesenteric trunk: a case report and review of literature. Vasc Specialist Int 2018;34:44-47.
- Choi C, Jung IM, Min SK, Han A, Ha J, Min SI. Inadvertent vascular injury of the aorta or vena cava caused by acupuncture. J Vasc Surg Cases 2015;1:13-15.