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Weitzen SD, Nguyen Lam NT, Sanchez J. Scrotal and penile edema in a patient with incomplete kawasaki disease: a case report and brief literature review. BMC Pediatr 2024; 24:640. [PMID: 39385164 PMCID: PMC11462718 DOI: 10.1186/s12887-024-05099-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/15/2023] [Accepted: 09/23/2024] [Indexed: 10/11/2024] Open
Abstract
BACKGROUND Kawasaki disease (KD) is a medium artery vasculitis that predominantly affects children under age 5. Prompt diagnosis and treatment with IVIG and moderate dose aspirin is required to prevent the formation of coronary artery aneurysms. While scrotal edema and erythema have been seen in KD, here we present a distinctive case of incomplete Kawasaki with these features as well as penile edema. CASE PRESENTATION A 2-year-old, unvaccinated, African American male presented with 4 days of fever, bilateral limbic sparing conjunctivitis, a papular rash, unilateral shotty cervical lymphadenopathy, mild right-hand edema, and scrotal and penile edema and erythema. His labs were significant for sterile pyuria, elevated ALT, anemia for age, and hypoalbuminemia. He was diagnosed with incomplete Kawasaki disease and was treated with IVIG and moderate dose aspirin. Echocardiogram was negative for coronary aneurysms. His symptoms resolved and he was discharged home with low dose aspirin. At his 2-week follow up, he remained well-appearing with no refractory Kawasaki symptoms. CONCLUSION This is a unique case of penile edema in KD which to our knowledge has not been previously reported in literature. An understanding of genitourinary symptoms in Kawasaki disease can help timely diagnosis and treatment of the disease.
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Affiliation(s)
- Samuel David Weitzen
- Wayne State University School of Medicine, 540 E Canfield Street, Detroit, MI, 48201, USA.
| | - Nam Tran Nguyen Lam
- Wayne State University School of Medicine, 540 E Canfield Street, Detroit, MI, 48201, USA
| | - Joselito Sanchez
- Department of Pediatric Infectious Disease, Children's Hospital of Michigan, 3901 Beaubien Blvd, Detroit, MI, 48201, USA
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Zhang S, Wang Q, Li Z, Guo Q. Testicular ischemia associated with IgA vasculitis in a child: a case report and literature review. Front Pediatr 2023; 11:1219878. [PMID: 37635796 PMCID: PMC10450908 DOI: 10.3389/fped.2023.1219878] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/09/2023] [Accepted: 07/26/2023] [Indexed: 08/29/2023] Open
Abstract
Testicular necrosis is a rare and severe complication of immunoglobulin A (IgA) vasculitis (IgAV). Herein, We report a case of a 10-year-old boy who was admitted to the hospital due to skin purpura and intermittent abdominal pain for 10 days and bilateral testicular pain for 2 days. Scrotal ultrasonography indicated right testicle ischemia, right epididymo-orchitis, and bilateral hydrocele of the testis. Scrotal surgical exploration revealed significant swelling and darkening of the right testicle. Conservative treatment led to improvement in his condition, and he was discharged. During 3 months of follow-up, there was no recurrence of skin purpura or pain, and the urine tests were normal. Color ultrasound indicated only partial blood flow signal to the right testicle tissue, which was slightly smaller than the left testicle. This case highlights the need for continuous attention from clinicians to the signs and symptoms of the reproductive system during the diagnosis and treatment of IgAV. Continuous monitoring with ultrasound can aid in early detection, diagnosis, and treatment of reproductive system lesions of IgA vasculitis.
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Affiliation(s)
- Shuya Zhang
- The First Clinical Medical College, Henan University of Chinese Medicine, Zhengzhou, China
| | - Qingwen Wang
- The First Clinical Medical College, Henan University of Chinese Medicine, Zhengzhou, China
| | - Ziwei Li
- The First Clinical Medical College, Henan University of Chinese Medicine, Zhengzhou, China
| | - Qingyin Guo
- Department of Pediatrics, The First Affiliated Hospital of Henan University of Chinese Medicine, Zhengzhou, China
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Xu YY, Huang XB, Wang YG, Zheng LY, Li M, Dai Y, Zhao S. Development of Henoch-Schoenlein purpura in a child with idiopathic hypereosinophilia syndrome with multiple thrombotic onset: A case report. World J Clin Cases 2023; 11:952-961. [PMID: 36818609 PMCID: PMC9928698 DOI: 10.12998/wjcc.v11.i4.952] [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: 11/16/2022] [Revised: 12/11/2022] [Accepted: 01/09/2023] [Indexed: 02/03/2023] Open
Abstract
BACKGROUND The incidence of pulmonary embolism (PE) in children is low, but its mortality is high. Hypereosinophilic syndrome (HES) is a group of diseases caused by an abnormal increase in eosinophilic granulocytes resulting in multiple-organ dysfunction. The urgent event of thromboembolism in the pulmonary region provoked by eosinophils in idiopathic HES (IHES) is relatively unusual. This article reports a case of IHES with multiple PEs and left leg venous thrombosis as the first manifestation. One month later, the patient developed Henoch-Schonlein purpura (HSP), which is very rare.
CASE SUMMARY We report the case of a 12-year-old boy who was admitted to the hospital with dyspnea, left leg pain, and aggravation. He had bilateral PE and left leg venous embolism with mild eosinophilia. Low-molecular-weight heparin and urokinase were given. At the same time, the interventional department was contacted about filter implantation, followed by urokinase thrombolysis. The left leg thrombus was aspirated under ultrasound guidance. He was discharged from the hospital on rivaroxaban. One month later, he developed a rash on both legs and ankle pain consistent with HSP, with severe eosinophilia and motor and sensory disturbances. The patient was diagnosed with IHES with multiple embolisms complicated by HSP after excluding other causes of the eosinophil elevation. After glucocorticoid treatment, the symptoms were relieved, but the patient later developed purpura nephritis.
CONCLUSION We report a rare and life-threatening case of IHES with multiple embolisms associated with HSP. A mild elevation of eosinophils early in the disease leads to difficulties in diagnosis and delayed treatment.
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Affiliation(s)
- Yan-Yan Xu
- Department of Pediatric Cardiovascular, Anhui Province Children's Hospital, Anhui Hospital of Children's Hospital Affiliated with Fudan University, Hefei 230051, Anhui Province, China
| | - Xiao-Bi Huang
- Department of Pediatric Cardiovascular, Anhui Province Children's Hospital, Anhui Hospital of Children's Hospital Affiliated with Fudan University, Hefei 230051, Anhui Province, China
| | - Yun-Gong Wang
- Department of Pediatric Cardiovascular, Anhui Province Children's Hospital, Anhui Hospital of Children's Hospital Affiliated with Fudan University, Hefei 230051, Anhui Province, China
| | - Li-Yun Zheng
- Department of Pediatric Cardiovascular, Anhui Province Children's Hospital, Anhui Hospital of Children's Hospital Affiliated with Fudan University, Hefei 230051, Anhui Province, China
| | - Min Li
- Department of Pediatric Intensive Care Unit, Anhui Province Children's Hospital, Anhui Hospital of Children's Hospital affiliated with Fudan University, Hefei 230051, Anhui Province, China
| | - Yu Dai
- Department of Pediatrics, The Fourth Affiliated Hospital of Anhui Medical University, Hefei 230032, Anhui Province, China
| | - Sheng Zhao
- Department of Pediatric Cardiovascular, Anhui Province Children's Hospital, Anhui Hospital of Children's Hospital Affiliated with Fudan University, Hefei 230051, Anhui Province, China
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Montorfani-Janett VML, Montorfani GE, Lavagno C, Gualco G, Bianchetti MG, Milani GP, Lava SAG, Cristallo Lacalamita M. External Male Genitalia in Henoch-Schönlein Syndrome: A Systematic Review. CHILDREN (BASEL, SWITZERLAND) 2022; 9:children9081154. [PMID: 36010045 PMCID: PMC9406875 DOI: 10.3390/children9081154] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 06/27/2022] [Revised: 07/16/2022] [Accepted: 07/26/2022] [Indexed: 11/23/2022]
Abstract
The external genitalia are notoriously implicated in every fifth male with Henoch−Schönlein syndrome. Nonetheless, the underlying conditions are poorly categorized. To characterize the involvement of the external male genitalia in this vasculitis, we performed a systematic review of the literature. For the final analysis, we selected 85 reports published between 1972 and 2022, which reported on 114 Henoch−Schönlein cases (≤ 18 years, N = 104) with a penile (N = 18), a scrotal (N = 77), or both a penile and a scrotal (N = 19) involvement. The genital involvement mostly appeared concurrently with or after the cutaneous features of Henoch−Schönlein syndrome, while it preceded the presentation of Henoch−Schönlein syndrome in 10 cases. Patients with penile involvement (N = 37) presented with swelling (N = 26), erythema (N = 23), and purpuric rash (N = 15). Most patients were otherwise asymptomatic except for transient micturition disorders (N = 2) or priapism (N = 2). Patients with scrotal involvement (N = 96) presented with pain (N = 85), swelling (N = 79), erythema (N = 42), or scrotal purpura (N = 22). The following scrotal structures were often involved: scrotal skin (N = 83), epididymis (N = 49), and testes (N = 39). An ischemic testicular damage was noted in nine patients (four with torsion and five without). The scrotal skin involvement was mostly bilateral, while that of the epididymis and testis were mostly (p < 0.0001) unilateral (with a significant predilection for the left side). In conclusion, this analysis allows for better categorization of the involvement of external male genitalia in Henoch−Schönlein vasculitis. Scrotal involvement can result from skin inflammation, epididymitis, orchitis, or testicular ischemia.
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Affiliation(s)
- Valentina M. L. Montorfani-Janett
- Family Medicine, Faculty of Biomedical Sciences, Università della Svizzera Italiana, 6900 Lugano, Switzerland; (V.M.L.M.-J.); (G.E.M.); (M.G.B.)
| | - Gabriele E. Montorfani
- Family Medicine, Faculty of Biomedical Sciences, Università della Svizzera Italiana, 6900 Lugano, Switzerland; (V.M.L.M.-J.); (G.E.M.); (M.G.B.)
| | - Camilla Lavagno
- Pediatric Emergency Department, University Children’s Hospital Zurich, 8032 Zurich, Switzerland;
| | - Gianluca Gualco
- Pediatric Institute of Southern Switzerland, Ente Ospedaliero Cantonale, 6500 Bellinzona, Switzerland;
- Faculty of Biomedical Sciences, Università della Svizzera Italiana, 6900 Lugano, Switzerland
| | - Mario G. Bianchetti
- Family Medicine, Faculty of Biomedical Sciences, Università della Svizzera Italiana, 6900 Lugano, Switzerland; (V.M.L.M.-J.); (G.E.M.); (M.G.B.)
- Faculty of Biomedical Sciences, Università della Svizzera Italiana, 6900 Lugano, Switzerland
| | - Gregorio P. Milani
- Pediatric Unit, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, 20122 Milan, Italy;
- Department of Clinical Sciences and Community Health, Università degli Studi di Milano, 20122 Milan, Italy
| | - Sebastiano A. G. Lava
- Pediatric Cardiology Unit, Department of Pediatrics, Centre Hospitalier Universitaire Vaudois and University of Lausanne, 1011 Lausanne, Switzerland
- Heart Failure and Transplantation, Department of Paediatric Cardiology, Great Ormond Street Hospital, London WC1N 3JH, UK
- Correspondence:
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Castro-Avendaño JL, Rosero-Rosero FD, Donoso-Donoso W, Cagua-Agudelo JE, Rivillas-Reyes JF, Rey-Melendez DDP. Trombosis de la vena espermática derecha. Primer caso clínico reportado en Colombia. CASE REPORTS 2021. [DOI: 10.15446/cr.v7n2.87345] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/09/2022] Open
Abstract
Introducción. La trombosis de la vena espermática (TVE) es una entidad muy poco frecuente de la cual no se tienen datos epidemiológicos específicos. Su presentación es de predominio izquierdo, en la mayoría de los casos su causa es desconocida y su diagnóstico suele realizarse por exclusión en el estudio del escroto agudo. A continuación, se presenta el primer caso documentado de TVE en Colombia y uno de los pocos de lateralidad derecha en el mundo.
Presentación del caso. Hombre de 21 años quien consultó al servicio de urgencias de una institución de tercer nivel de atención de Bogotá, Colombia, por un cuadro clínico de una semana de evolución consistente en dolor en región inguinoescrotal derecha que aumentó progresivamente de intensidad y estuvo acompañado de un pico febril no cuantificado; el paciente indicó como único antecedente médico un trauma craneoencefálico leve sin secuelas a los 20 años. Se realizó estudio con ecografía Doppler testicular en donde se observó trombosis del cordón espermático derecho. Se inició manejó con antibioticoterapia, heparinas de bajo peso molecular y cumarínicos, con lo cual se obtuvo una adecuada respuesta y la resolución del cuadro.
Conclusión. La TVE representa un reto diagnóstico, por lo que se requiere de una alta sospecha clínica, la cual se genera teniendo conocimiento de la entidad e incluyéndola en el diagnóstico diferencial del escroto agudo. La ecografía Doppler es una herramienta fundamental para diagnosticar TVE, además permite iniciar un tratamiento conservador oportuno, seguro y eficaz que evite desenlaces desfavorables para el paciente, lo que resulta ser costo-efectivo.
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Du L, Wang P, Liu C, Li S, Yue S, Yang Y. Multisystemic manifestations of IgA vasculitis. Clin Rheumatol 2020; 40:43-52. [PMID: 32557258 DOI: 10.1007/s10067-020-05166-5] [Citation(s) in RCA: 32] [Impact Index Per Article: 6.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/19/2020] [Revised: 05/07/2020] [Accepted: 05/11/2020] [Indexed: 01/27/2023]
Abstract
Immunoglobulin A vasculitis (IgAV), also known as Henoch-Schönlein Purpura, is one of the most common kind of systemic vasculitis in children, and due to the involvement of small blood vessels throughout the body, this disease can cause a variety of symptoms in different organs. Our aim was to review the data on various systemic manifestations of IgAV. A research of the literature was performed in PubMed database, utilizing the MeSH terms "IgA vasculitis" and "Henoch Schönlein Purpura". According to the predetermined structure of the manuscript, we extracted and sorted out the relevant data. Clinically, almost all the patients will present with palpable skin purpura, together with arthritis, gastrointestinal tract involvement, or kidney damage. Other rare systemic manifestations include neurological symptoms, scrotal involvement, and cardiopulmonary disease. When uncommon complications occur, patients may be misdiagnosed as other diseases, thus delaying treatment. Although the course of IgAV is mostly self-limited, misdiagnosis can also lead to a poor prognosis. A comprehensive awareness to the clinical manifestations of IgAV is the necessary prerequisite for its timely diagnosis. Prompt diagnosis and adequate treatment are essential for optimal results.
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Affiliation(s)
- Lina Du
- Department of Chinese Medicine, National Center for Children's Health, Beijing Children's Hospital, Capital Medical University, No. 56 Nanlishi Road, Beijing, 100045, China
| | - Panpan Wang
- Department of Chinese Medicine, National Center for Children's Health, Beijing Children's Hospital, Capital Medical University, No. 56 Nanlishi Road, Beijing, 100045, China
| | - Chang Liu
- Department of Chinese Medicine, National Center for Children's Health, Beijing Children's Hospital, Capital Medical University, No. 56 Nanlishi Road, Beijing, 100045, China
| | - Shaojing Li
- Department of Chinese Medicine, National Center for Children's Health, Beijing Children's Hospital, Capital Medical University, No. 56 Nanlishi Road, Beijing, 100045, China
| | - Shuang Yue
- Department of Chinese Medicine, National Center for Children's Health, Beijing Children's Hospital, Capital Medical University, No. 56 Nanlishi Road, Beijing, 100045, China
| | - Yan Yang
- Department of Chinese Medicine, National Center for Children's Health, Beijing Children's Hospital, Capital Medical University, No. 56 Nanlishi Road, Beijing, 100045, China.
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Right Testicular Infarction Secondary to Spontaneous Testicular Vein Thrombosis in a Child: Case Report and Literature Review. Urology 2019; 130:144-147. [DOI: 10.1016/j.urology.2019.03.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/10/2018] [Revised: 03/09/2019] [Accepted: 03/12/2019] [Indexed: 10/27/2022]
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Tracy A, Subramanian A, Adderley NJ, Cockwell P, Ferro C, Ball S, Harper L, Nirantharakumar K. Cardiovascular, thromboembolic and renal outcomes in IgA vasculitis (Henoch-Schönlein purpura): a retrospective cohort study using routinely collected primary care data. Ann Rheum Dis 2019; 78:261-269. [PMID: 30487151 DOI: 10.1136/annrheumdis-2018-214142] [Citation(s) in RCA: 43] [Impact Index Per Article: 7.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/18/2018] [Revised: 10/30/2018] [Accepted: 11/01/2018] [Indexed: 01/27/2023]
Abstract
BACKGROUND IgA vasculitis (IgAV, Henoch-Schönlein purpura) is a small-vessel vasculitis most common in children but also occurring in adults. Case series have suggested that IgAV may be associated with cardiovascular disease and venous thromboembolism, but this has not been evaluated in population-based studies. Renal disease and hypertension are possible complications of the disease with unknown incidence. METHODS Using a large UK primary care database, we conducted an open retrospective matched cohort study of cardiovascular, venous thrombotic and renal outcomes in adult-onset and childhood-onset IgAV. Control participants were selected at a 2:1 ratio, matched for age and sex. Adjusted HRs (aHRs) were calculated using Cox proportional hazards models. RESULTS 2828 patients with adult-onset IgAV and 10 405 patients with childhood-onset IgAV were compared with age-matched and sex-matched controls. There was significantly increased risk of hypertension (adult-onset aHR 1.42, 95% CI 1.19 to 1.70, p < 0.001; childhood-onset aHR 1.52, 95% CI 1.22 to 1.89, p < 0.001) and stage G3-G5 chronic kidney disease (adult-onset aHR 1.54, 95% CI 1.23 to 1.93, p < 0.001; childhood-onset aHR 1.89, 95% CI 1.16 to 3.07, p=0.010). There was no evidence of association with ischaemic heart disease, cerebrovascular disease or venous thromboembolism. All-cause mortality was increased in the adult-onset IgAV cohort compared with controls (aHR 1.27, 95% CI 1.07 to 1.50, p=0.006). CONCLUSIONS Patients with IgAV are at increased risk of hypertension and chronic kidney disease (CKD) compared with individuals without IgAV; analysis restricted to adult-onset IgAV patients showed increased mortality. Appropriate surveillance and risk factor modification could improve long-term outcomes in these patients.
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Affiliation(s)
- Alexander Tracy
- Centre for Translational Inflammation Research, Institute of Clinical Sciences, University of Birmingham, Birmingham, UK
| | | | - Nicola J Adderley
- Institute of Applied Health Research, University of Birmingham, Birmingham, UK
| | - Paul Cockwell
- Department of Renal Medicine, University Hospitals Birmingham NHS Trust, Birmingham, UK
| | - Charles Ferro
- Department of Renal Medicine, University Hospitals Birmingham NHS Trust, Birmingham, UK
| | - Simon Ball
- Department of Renal Medicine, University Hospitals Birmingham NHS Trust, Birmingham, UK
| | - Lorraine Harper
- Centre for Translational Inflammation Research, Institute of Clinical Sciences, University of Birmingham, Birmingham, UK
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De Maddi F, Sottile R, Andreozzi L, Rigante D. Deep thrombophlebitis masked by peripheral arthritis in Henoch-Schönlein purpura. Int J Rheum Dis 2016; 19:1357-1358. [PMID: 25496261 DOI: 10.1111/1756-185x.12554] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/28/2022]
Affiliation(s)
- Francesco De Maddi
- Department of Pediatrics, Santobono-Pausilipon Children Hospital, Naples, Italy
| | - Rita Sottile
- Department of Pediatrics, Santobono-Pausilipon Children Hospital, Naples, Italy
| | - Laura Andreozzi
- Institute of Pediatrics, Università Cattolica Sacro Cuore, Rome, Italy
| | - Donato Rigante
- Institute of Pediatrics, Università Cattolica Sacro Cuore, Rome, Italy
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Dalpiaz A, Schwamb R, Miao Y, Gonka J, Walzter W, Khan SA. Urological Manifestations of Henoch-Schonlein Purpura: A Review. Curr Urol 2015; 8:66-73. [PMID: 26889120 DOI: 10.1159/000365692] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/16/2014] [Accepted: 09/30/2014] [Indexed: 11/19/2022] Open
Abstract
Henoch-Schonlein purpura (HSP) is an immune-mediated systemic vasculitis generally found in children. The standard manifestations of HSP are palpable purpura, arthritis, abdominal pain, and renal complications. Although less common, there are significant urological manifestations associated with HSP. The primary objective of this review is to encourage better understanding and management of HSP by emphasizing the common and rare manifestations of HSP, how they are diagnosed, and the latest treatment options for mild to severe complications. Medline searches of HSP and its urological manifestations were conducted along with searches on current diagnostic and treatment methods. Urological manifestations of HSP involve the kidney, ureter, bladder, prostate, scrotum, testicle, and penis. Diagnosis and management of HSP are not always clear due to differential diagnosis and diversity of symptom presentation. Treatment for HSP is mainly supportive and includes use of nonsteroidal anti-inflammatory drugs for pain relief. In more severe cases, glucocorticoids, methylprednisolone, plasmapheresis, and peritoneal and hemodialysis are reported successful. It is important to note different symptoms of HSP in order to distinguish HSP from other diseases. Early diagnosis may prevent severe complications. Treatment options vary from conservative to invasive depending on the severity of the disease and time frame of diagnosis.
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Affiliation(s)
- Amanda Dalpiaz
- Department of Physiology and Biophysics, Stony Brook, N.Y., USA
| | - Richard Schwamb
- Department of Physiology and Biophysics, Stony Brook, N.Y., USA
| | - Yimei Miao
- Department of Urology, SUNY School of Medicine, Stony Brook University, Stony Brook, N.Y., USA
| | - Jacquelyn Gonka
- Department of Urology, SUNY School of Medicine, Stony Brook University, Stony Brook, N.Y., USA
| | - Wayne Walzter
- Department of Urology, SUNY School of Medicine, Stony Brook University, Stony Brook, N.Y., USA
| | - Sardar A Khan
- Department of Urology, SUNY School of Medicine, Stony Brook University, Stony Brook, N.Y., USA
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Peracha J, Morgan MD. Urological manifestations and treatment of the primary systemic vasculitides. World J Clin Urol 2015; 4:5-20. [DOI: 10.5410/wjcu.v4.i1.5] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/28/2014] [Revised: 11/17/2014] [Accepted: 12/31/2014] [Indexed: 02/06/2023] Open
Abstract
The primary systemic vasculitides (PSV) are a group of rare inflammatory disorders affecting blood vessels of varying size and multiple organs. Urological manifestations of PSV are uncommon. Testicular vasculitis is the most commonly reported finding and is associated with Polyarteritis Nodosa (PAN), Henoch-Schönlein Purpura (HSP), anti-neutrophil cytoplasm antibody associated Vasculitides (AAV), Giant Cell Arteritis (GCA) and Kawasaki disease. Prostatic vasculitis has been reported in association with GCA and AAV. Ureteric involvement has been noted in PAN, HSP and AAV. Other urogenital manifestations of PSV include genital ulceration and bladder dysfunction in Behçets Disease and haematuria which is commonly seen in many of the PSV. Finally, therapies used to treat the PSV, especially cyclophosphamide, are associated with urological side-effects including haemorrhagic cystitis and urothelial malignancy. The aim of this review is to examine how the urological system is involved in the PSV. Each PSV is examined in turn, with a brief clinical description of the disease followed by a description of the urological manifestations and management. Identification of urological manifestations of PSV is important as in many cases symptoms may improve with immunosuppressive therapy, avoiding the need for invasive surgery. Additionally, patients who present with isolated urogenital PSV are at higher risk of developing subsequent systemic vasculitis and will need to be followed up closely.
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12
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Zaidi AU, Berman B. Crossing the thrombotic threshold: deep vein thrombosis in Henoch-Schönlein purpura. Clin Pediatr (Phila) 2014; 53:1396-8. [PMID: 24634422 DOI: 10.1177/0009922814526983] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Affiliation(s)
- Ahmar U Zaidi
- Oakland University William Beaumont School of Medicine, Royal Oak, MI, USA
| | - Brian Berman
- Oakland University William Beaumont School of Medicine, Royal Oak, MI, USA
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Li L, Zhang J, Zhang Y, Ji H. Thrombosis warning in children suffering from henoch-schonlein purpura. Indian J Dermatol 2013; 58:409. [PMID: 24082216 PMCID: PMC3778811 DOI: 10.4103/0019-5154.117349] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022] Open
Abstract
Henoch-Schonlein Purpura (HSP) is one of the most common types of vasculitis in children. The characteristic clinical manifestations include non-thrombocytopenic purpura, arthritis or arthralgia, abdominal pain, gastrointestinal hemorrhage and renal abnormalities. Thrombosis has been reported as, a rare complication of HSP. We present the case of a 14-year-old boy who was diagnosed with HSP and suspected superior mesenteric vein thrombosis. We reviewed the relevant literature and found eight similar reported cases. HSP is associated with thrombosis and HSP itself and some risk factors may result in thrombosis. We suggest that physicians should monitor patients with HSP who are at a higher risk of developing thrombosis more closely.
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Affiliation(s)
- Luan Li
- Department of Dermatology and Venereology, First Affiliated Hospital, Dalian Medical University, Dalian, China
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Fukuda S, Takahashi T, Kumori K, Takahashi Y, Yasuda K, Kasai T, Yamaguchi S. Idiopathic testicular infarction in a boy initially suspected to have acute epididymo-orchitis associated with mycoplasma infection and Henoch-Schönlein purpura. J Pediatr Urol 2009; 5:68-71. [PMID: 18753011 DOI: 10.1016/j.jpurol.2008.07.003] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/12/2008] [Accepted: 07/14/2008] [Indexed: 10/21/2022]
Abstract
Idiopathic testicular infarction without torsion of spermatic cord is a rare condition. We present a 12-year-old boy originally suspected of acute epididymo-orchitis, but subsequently an orchiectomy was necessary owing to unpredicted testicular infarction not associated with torsion. Elevation of immunoglobulin M against mycoplasma, reduction in serum factor XIII and the presence of sufficient blood flow detected by color Doppler ultrasonography upon initial manifestation suggested that the boy was affected by epididymo-orchitis associated with Henoch-Schönlein purpura or mycoplasma infection. However, progressive testicular enlargement was observed and subsequently testicular blood flow became barely detectable. Our case indicates that the presence of sufficient blood flow upon initial diagnosis in the affected testis does not necessarily exclude infarction and continuous monitoring of blood flow may be required to avoid radical orchiectomy.
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Affiliation(s)
- Seiji Fukuda
- Department of Pediatrics, Shimane University School of Medicine, Izumuo, Japan.
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Castillo OA, Diaz M, Vitagliano GJ, Metrebian E. Pulmonary thromboembolism secondary to left spermatic vein thrombosis: a case report. Urol Int 2008; 80:217-8. [PMID: 18362496 DOI: 10.1159/000112617] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/30/2006] [Accepted: 07/21/2006] [Indexed: 11/19/2022]
Abstract
Spermatic vein thrombosis is a particularly rare entity which can be difficult to diagnose. Pulmonary embolism associated with spermatic vein thrombosis is rarely seen. We report the diagnosis and management of a case at our institution and recommend spermatic vein ligation as the definite treatment for thrombosed spermatic veins associated with pulmonary thromboembolism. We prefer laparoscopy as a minimally invasive approach because of its clear advantages over open surgery.
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Affiliation(s)
- Octavio A Castillo
- Section of Endourology and Laparoscopic Urology, Department of Urology, Clínica Santa María, Santiagode Chile, Chile.
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16
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Jequier M, Thurler-Thiele F, Lepori D, Gehri M. [Radiological quiz of the month. A case of a swollen and painful testis in a child]. Arch Pediatr 2005; 12:1513-4, 1500. [PMID: 16122918 DOI: 10.1016/j.arcped.2005.06.020] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/20/2005] [Accepted: 06/30/2005] [Indexed: 11/17/2022]
Affiliation(s)
- M Jequier
- Département médico-chirurgical de pédiatrie, hôpital de l'Enfance de Lausanne, ch. de Montétan 16, 1000 Lausanne 7, Suisse
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17
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Abstract
BACKGROUND/PURPOSE The aim of this study was to present the current experience and evidence relating to surgical evaluation and management of nonrenal genitourinary presentation in children with Henoch-Schönlein purpura (HSP). Henoch-Schönlein purpura is the most common systemic vasculitis in childhood. Presenting symptoms include a rash, arthralgia, abdominal pain, and, frequently, renal involvement. Genitourinary symptoms are infrequently reported; however, surgical evaluation is often required. METHODS A MEDLINE literature search of the last 3 decades was conducted using the terms Henoch-Schönlein purpura in combination with acute scrotum or genitourinary, genital, or related symptoms as the keywords. Reference lists of retrieved articles were reviewed for further relevant articles. Case reports were included when no larger case series were found. Articles of foreign language were included if abstracts proved sufficient information. RESULTS Genitourinary manifestations are mainly anecdotally reported. Scrotal pain and swelling were the most frequent nonrenal genitourinary symptoms reported in children with HSP, occurring in about 13% of boys evaluated for HSP. No prospective, population-based reports on genitourinary manifestations in HSP were found, thus making estimation of the true incidence difficult. CONCLUSIONS A wide variety of nonrenal genitourinary pathology may be expected by surgical evaluation of patients with HSP. Most cases are self-limited in nature. Scrotal symptoms are frequent, but testicular torsion is highly unlikely when the diagnosis of HSP is established. Clinical vigilance should be paid to patients with possible obstructive ureteritis, for whom prompt surgical intervention may be indicated.
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Affiliation(s)
- Kjetil Søreide
- Department of Surgery, Stavanger University Hospital, N-4068 Stavanger, Norway.
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Sari I, Akar S, Secil M, Birlik M, Kefi A, Onen F, Celebi I, Akkoc N. Thrombosis and priapism in a patient with Henoch-Schonlein purpura. Rheumatol Int 2004; 25:472-4. [PMID: 16133584 DOI: 10.1007/s00296-004-0532-3] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/01/2004] [Accepted: 08/13/2004] [Indexed: 10/26/2022]
Abstract
Henoch-Schönlein purpura (HSP) belongs to the category of systemic small-vessel vasculitis. Although long-term outcome is generally good, serious complications may occur. Thrombosis and priapism have been reported only as extremely rare complications of HSP. We describe a 37-year-old man who developed recurrent thrombotic events shortly after he had been diagnosed as having HSP. Although he had additional risk factors for thrombosis, such as prothrombin G20210A mutation and use of celecoxib before the last episode, temporal relation of the thrombotic attacks to the onset of HSP suggest that the disease itself may lead to a prothrombotic state. This case is the first adult HSP patient with priapism, which probably developed secondary to thrombosis of the dorsal penile vein.
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Affiliation(s)
- Ismail Sari
- Division of Rheumatology, Department of Internal Medicine, Dokuz Eylul University School of Medicine, 35340, Inciralti, Izmir, Turkey.
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Abstract
Inadequate understanding of the pathogenesis and etiology of vascular inflammation continues to hinder progress in the diagnosis and treatment of pediatric vasculitis. The greatest amount of work is being done in the most common vasculitides of childhood, including Kawasaki disease and Henoch-Schönlein purpura. Discussion of rarer types of vasculitis, on the other hand, such as antineutrophil cytoplasmic antibody-positive small vessel diseases, is largely restricted to case reports. Most aspects of the care of children with Wegener granulomatosis and microscopic polyangiitis are derived by extrapolating from data about adults. Virtually no data are available concerning ways in which these diseases may be different in children.
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Affiliation(s)
- A Yalcindag
- Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA
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