{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,8]],"date-time":"2026-05-08T00:24:02Z","timestamp":1778199842500,"version":"3.51.4"},"reference-count":25,"publisher":"MDPI AG","issue":"5","license":[{"start":{"date-parts":[[2021,5,12]],"date-time":"2021-05-12T00:00:00Z","timestamp":1620777600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Medicina"],"abstract":"<jats:p>Introduction: Leriche syndrome is an aortoiliac occlusive disease caused by atherosclerotic occlusion. We report a case of Leriche syndrome with a fracture that was suspected as complex regional pain syndrome (CRPS), as the post-traumatic pain gradually worsened in the form of excruciating neuropathic pain. Case Report: A 52-year-old woman with a history of hypertension was referred to the Department of Pain Medicine from a local orthopedic clinic because of suspected CRPS for excruciating neuropathic pain for one month. She complained of gait dysfunction and severe pain in the right foot following an incident of trauma with the right first toe. The average pain intensity assessed using the visual analog scale (VAS) was 90 (0: no pain, 100: the worst pain imaginable), and the neuropathic pain was evident as a score of 6\/10 on Douleur neuropathique 4. Allodynia, hyperalgesia, blue discoloration of the skin, asymmetric temperature change (1.38 \u00b0C), and edematous soft tissue changes were observed. Ultrasonography showed a chip fracture in the first distal phalanx of the right first toe. The diagnosis was most probably CRPS type I according to the Budapest research criteria for CRPS. However, multiple pain management techniques were insufficient in controlling the symptoms. A month and a half later, an ankle-brachial index score of less than 0.4 suggested severe peripheral artery disease. Computed tomography angiography showed total occlusion between the infrarenal abdominal aorta and the bilateral common iliac arteries. Therefore, she underwent aortic-bifemoral bypass surgery with a diagnosis of Leriche syndrome. Three months after the surgery, the average pain intensity was graded as 10 on the VAS (0\u2013100), the color of the skin of the right first toe improved and no gait dysfunction was observed. Conclusion: A chip fracture in a region with insufficient blood flow could manifest as excruciating neuropathic pain in Leriche syndrome.<\/jats:p>","DOI":"10.3390\/medicina57050486","type":"journal-article","created":{"date-parts":[[2021,5,12]],"date-time":"2021-05-12T22:46:14Z","timestamp":1620859574000},"page":"486","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Leriche Syndrome Misdiagnosed as Complex Regional Pain Syndrome in a Patient with Neuropathic Pain Caused by a Chip Fracture: A Case Report"],"prefix":"10.3390","volume":"57","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7085-179X","authenticated-orcid":false,"given":"Byeong-Cheol","family":"Lee","sequence":"first","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8064-9904","authenticated-orcid":false,"given":"Dae-Seok","family":"Oh","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hyun-Seong","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Se-Hun","family":"Kim","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jae-Hong","family":"Park","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ki-Hwa","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hyo-Joong","family":"Kim","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ji-Hyun","family":"Yang","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9029-4991","authenticated-orcid":false,"given":"Sang-Eun","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Anesthesiology and Pain Medicine, Haeundae Paik Hospital of Inje University, 875 Haeundae-ro, Haeundae-gu, Busan 48108, Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2021,5,12]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"223","DOI":"10.1007\/s12245-008-0039-x","article-title":"Leriche syndrome","volume":"1","author":"Lee","year":"2008","journal-title":"Int. J. Emerg. Med."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1102","DOI":"10.1007\/s11606-010-1412-z","article-title":"Leriche syndrome","volume":"25","author":"Frederick","year":"2010","journal-title":"J. Gen. Intern. Med."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"193","DOI":"10.1097\/00000658-194802000-00001","article-title":"The syndrome of thrombotic obliteration of the aortic bifurcation","volume":"127","author":"Leriche","year":"1948","journal-title":"Ann. Surg."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"861","DOI":"10.1056\/NEJMcp1507631","article-title":"Peripheral Artery Disease","volume":"374","author":"Kullo","year":"2016","journal-title":"N. Engl. J. Med."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"95","DOI":"10.1177\/1358863X13480001","article-title":"Methods of symptom evaluation and their impact on peripheral artery disease (PAD) symptom prevalence: A review","volume":"18","author":"Schorr","year":"2013","journal-title":"Vasc. Med."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"1008","DOI":"10.1212\/WNL.57.6.1008","article-title":"Chronic ischemic monomelic neuropathy from critical limb ischemia","volume":"57","author":"Weinberg","year":"2001","journal-title":"Neurology"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"471","DOI":"10.1002\/mus.10235","article-title":"Axonal neuropathy in chronic peripheral arterial occlusive disease","volume":"26","author":"Weber","year":"2002","journal-title":"Muscle Nerve Off. J. Am. Assoc. Electrodiagn. Med."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"125","DOI":"10.5758\/vsi.2014.30.4.125","article-title":"Prevalence and risk factors for the peripheral neuropathy in patients with peripheral arterial occlusive disease","volume":"30","author":"Kim","year":"2014","journal-title":"Vasc. Spec. Int."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"581","DOI":"10.1111\/tbj.13622","article-title":"Management of neuropathic pain: A graph theory-based presentation of literature review","volume":"26","author":"Coraci","year":"2020","journal-title":"Breast J."},{"key":"ref_10","doi-asserted-by":"crossref","unstructured":"Giovannini, S., Coraci, D., Brau, F., Galluzzo, V., Loreti, C., Caliandro, P., Padua, L., Maccauro, G., Biscotti, L., and Bernabei, R. (2021). Neuropathic Pain in the Elderly. Diagnostics, 11.","DOI":"10.3390\/diagnostics11040613"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"S35","DOI":"10.1177\/0003319797048005S04","article-title":"Patient Assessment and Examples Patient Assessment and Examples of a Method of Treatment","volume":"48","author":"Dillon","year":"1997","journal-title":"Angiology"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"375","DOI":"10.1055\/s-0032-1330431","article-title":"Manifestation of Acute Leriche Syndrome as Cauda Equina Syndrome","volume":"185","author":"Groth","year":"2013","journal-title":"Rofo"},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"132","DOI":"10.5535\/arm.2014.38.1.132","article-title":"Right calf claudication revealing leriche syndrome presenting as right sciatic neuropathy","volume":"38","author":"Seol","year":"2014","journal-title":"Ann. Rehabil. Med."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"2929","DOI":"10.1007\/s00198-017-4133-x","article-title":"Bone mineral density in adults with Down syndrome","volume":"28","author":"Liperoti","year":"2017","journal-title":"Osteoporos. Int."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"380","DOI":"10.1002\/mus.880180403","article-title":"Progression of neuropathy in peripheral arterial disease","volume":"18","author":"England","year":"1995","journal-title":"Muscle Nerve Off. J. Am. Assoc. Electrodiagn. Med."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"201","DOI":"10.1016\/j.pain.2008.03.027","article-title":"Characteristics of chronic ischemic pain in patients with peripheral arterial disease","volume":"139","author":"Irnich","year":"2008","journal-title":"Pain"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"708","DOI":"10.1067\/mva.2001.112329","article-title":"The value of toe pulse waves in determination of risks for limb amputation and death in patients with peripheral arterial disease and skin ulcers or gangrene","volume":"33","author":"Carter","year":"2001","journal-title":"J. Vasc. Surg."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"1257","DOI":"10.1016\/S0140-6736(01)06351-6","article-title":"Peripheral arterial disease","volume":"358","author":"Ouriel","year":"2001","journal-title":"Lancet"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"291","DOI":"10.1007\/s10456-017-9541-1","article-title":"The role of vasculature in bone development, regeneration and proper systemic functioning","volume":"20","author":"Filipowska","year":"2017","journal-title":"Angiogenesis"},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"311","DOI":"10.4248\/BR201304002","article-title":"Skeletal Blood Flow in Bone Repair and Maintenance","volume":"1","author":"Tomlinson","year":"2013","journal-title":"Bone Res."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"358","DOI":"10.1038\/nrrheum.2012.36","article-title":"Hypoxia-driven pathways in bone development, regeneration and disease","volume":"8","author":"Maes","year":"2012","journal-title":"Nat. Rev. Rheumatol."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"326","DOI":"10.1111\/j.1526-4637.2006.00169.x","article-title":"Proposed new diagnostic criteria for complex regional pain syndrome","volume":"8","author":"Harden","year":"2007","journal-title":"Pain Med."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"649","DOI":"10.1111\/j.1468-1331.2010.02947.x","article-title":"Complex regional pain syndromes: New pathophysiological concepts and therapies","volume":"17","author":"Seifert","year":"2010","journal-title":"Eur. J. Neurol."},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"165","DOI":"10.3344\/kjp.2017.30.3.165","article-title":"Bone scintigraphy in patients with pain","volume":"30","author":"Shin","year":"2017","journal-title":"Korean J. Pain"},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"1321.e5","DOI":"10.1016\/j.avsg.2013.12.013","article-title":"Aortoiliac occlusive disease presenting as sudden onset paraplegia","volume":"28","author":"Lai","year":"2014","journal-title":"Ann. Vasc. Surg."}],"container-title":["Medicina"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/1648-9144\/57\/5\/486\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T05:59:53Z","timestamp":1760162393000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/1648-9144\/57\/5\/486"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,5,12]]},"references-count":25,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2021,5]]}},"alternative-id":["medicina57050486"],"URL":"https:\/\/doi.org\/10.3390\/medicina57050486","relation":{},"ISSN":["1648-9144"],"issn-type":[{"value":"1648-9144","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,5,12]]}}}