Venous malformation as a rare cause of sciatic neuropathy
https://doi.org/10.46563/2686-8997-2023-4-3-175-180
EDN: qyuwmr
Abstract
Sciatic neuropathy is rare and difficult to diagnose in pediatrics and often associated with difficult to control neuropathic pain. The most common causes of sciatic nerve damage in children include trauma and iatrogenic lesions, less often the cause is tumor and vascular formations, among which intraneural venous malformations are the rarest. We present a clinical case of a 13-year boy suffered from right sciatic pain neuropathy with unexpressed neuropathic pain syndrome, asymmetry of the feet, scant impairment of motor and sensory function of the limb for five years, which changed the gait. Neurophysiological testing showed possible location of the process, an asymmetry of sensory and motor responses when testing the nerves of the legs, and abnormal EMG in sciatic-innervated muscles. The magnetic resonance imaging (MRI) showed focal enlargement of the right sciatic nerve in the pelvic region with enhancement upon administration of contrast. Due to the presence of pain in the limb and its forced position — bent at the knee joint, surgical treatment — endoneural neurolysis in combination with targeted therapy by immunosuppressant, was chosen as a therapeutic strategy. The histological study of the intraneural formation confirmed the presence of a venous malformation (VM) in the form of heterogeneous thick– and thin-walled vascular cavities by the presence of fibrosis and lymphocytic infiltration. The result of the treatment was the relief of neuropathic pain syndrome and the recovering of the child gait.
Compliance with ethical standards. Voluntary informed consent was obtained from the patient’s legal representative for treatment and publication of data.
Contribution:
Druzhinina E.S. — review of publications on the topic of the article, collection and analysis of data, writing the text of the manuscript;
Isaev I.V. — performing a surgical intervention, writing the text of an article;
Kostylev F.A. — data collection, writing the text of article;
Narbutov A.G. — performing a surgical intervention, writing the text of an article;
Zavadenko N.N. — writing the text of the article, checking critical intellectual content, final approval for publication of the manuscript.
All co-authors are responsible for the integrity of all parts of the manuscript and approval of its final version.
Acknowledgements. The study had no sponsorship.
Conflict of interest. The authors declare no conflict of interest.
Received: March 13, 2023
Accepted: March 27, 2023
Published: October 13, 2023
About the Authors
Evgeniya S. DruzhininaRussian Federation
Department of neurology, neurosurgery and medical genetics, Pediatric Faculty N.I. Pirogov Russian National Research Medical University; Department of nervous system diseases with course of medical genetics and pediatric neurology, Moscow, 117997, Russian Federation.
e-mail: umovaes@gmail.com
Igor V. Isaev
Russian Federation
Fedor A. Kostylev
Russian Federation
Anton G. Narbutov
Russian Federation
Nikolay N. Zavadenko
Russian Federation
References
1. Greene A.K., Liu A.S., Mulliken J.B., Chalache K., Fishman S.J. Vascular anomalies in 5,621 patients: guidelines for referral. J. Pediatr. Surg. 2011; 46(9): 1784–9. https://doi.org/10.1016/j.jpedsurg.2011.05.006
2. Prasad N.K., Chawla A., Lalezari S., de Ruiter G.C., Howe B.M., Amrami K.K., et al. A radiological classification system for intraneural vascular anomalies: assessment of potential for resection with high-resolution MRI. Acta. Neurochir. (Wien). 2016; 158(2): 329–34; discussion 334. https://doi.org/10.1007/s00701-015-2663-6
3. Khandelwal D.C., Sharma C.M., Srinivasan J. Pediatric sciatic neuropathies: a 30-year prospective study. Neurology. 2011; 77(19): 1772; author reply 1772. https://doi.org/10.1212/WNL.0b013e318239beb3
4. Jaque-Almendras C., Escobar R.G., Caicedo-Feijoo A., Beytía-Reyes M.L.A., Correa-Pérez S., Gejman-Enríquez R., et al. Pediatric sciatic neuropathy: Clinical presentation and long term follow up. Rev. Chil. Pediatr. 2020; 91(1): 85–93. https://doi.org/10.32641/rchped.v91i1.1355
5. Johal K.S., Platsas L., Chen H.C. Chronic sciatic nerve compression secondary to arteriovenous malformation: case discussion and literature review. Ann. R. Coll. Surg. Engl. 2021; 103(9): e278–81. https://doi.org/10.1308/rcsann.2020.7134
6. Van Gompel J.J., Griessenauer C.J., Scheithauer B.W., Amrami K.K., Spinner R.J. Vascular malformations, rare causes of sciatic neuropathy: a case series. Neurosurgery. 2010; 67(4): 1133–42; discussion 1142. https://doi.org/10.1227/NEU.0b013e3181ecc84e
7. Ilhanli I., Keskin O., Arslan E., Ekiz M. Sciatic nerve hypertrophy with Klippel-Trenaunay syndrome: a case report. Turk. Neurosurg. 2015; 25(3): 500–2. https://doi.org/10.5137/1019-5149.JTN.10523-14.1
8. Das R., Kumar I., Verma A., Shukla R.C. Spectrum of imaging findings in Klippel-Trenaunay syndrome affecting lower limbs: a report of three cases. Egypt. J. Radiol. Nucl. Med. 2019; 50: 104. https://doi.org/10.1186/s43055-019-0123-7
9. Pendleton C., Lenartowicz K.A., Howe B.M., Spinner R.J. Limb undergrowth in intraneural perineuriomas: an under-recognized association. World Neurosurg. 2020; 141: e670–6. https://doi.org/10.1016/j.wneu.2020.05.280
10. Mauermann M.L., Amrami K.K., Kuntz N.L., Spinner R.J., Dyck P.J., Bosch E.P., et al. Longitudinal study of intraneural perineurioma a benign, focal hypertrophic neuropathy of youth. Brain. 2009; 132(Pt. 8): 2265–76. https://doi.org/10.1093/brain/awp169
11. Brand C., Pedro M.T., Pala A., Heinen C., Scheuerle A., Braun M., et al. Perineurioma: A Rare Entity of Peripheral Nerve Sheath Tumors. J. Neurol. Surg. A. Cent. Eur. Neurosurg. 2022; 83(1): 1–5. https://doi.org/10.1055/s-0041-1726110
12. Srinivasan J., Escolar D., Ryan M., Darras B., Jones H.R. Pediatric sciatic neuropathies due to unusual vascular causes. J. Child. Neurol. 2008; 23(7): 738–41. https://doi.org/10.1177/0883073808314163
13. Cherian R.P., Li Y. Clinical and electrodiagnostic features of nontraumatic sciatic neuropathy. Muscle. Nerve. 2019; 59(3): 309–14. https://doi.org/10.1002/mus.26380
14. Uerschels A.K., Krogias C., Junker A., Sure U., Wrede K.H., Gembruch O. Modern treatment of perineuriomas: a case-series and systematic review. BMC Neurol. 2020; 20(1): 55. https://doi.org/10.1186/s12883-020-01637-z
15. Maruani A., Tavernier E., Boccara O., Mazereeuw-Hautier J., Leducq S., Bessis D., Guibaud L., Vabres P., Carmignac V., Mallet S., Barbarot S., Chiaverini C., Droitcourt C., Bursztejn A.C., Lengellé C., Woillard J.B., Herbreteau D., Le Touze A., Joly A., Léauté-Labrèze C., Powell J., Bourgoin H., Gissot V., Giraudeau B., Morel B. Sirolimus (Rapamycin) for Slow-Flow Malformations in Children: The Observational-Phase Randomized Clinical PERFORMUS Trial. JAMA Dermatol. 2021 Nov 1; 157(11): 1289-1298. https://doi.org/10.1001/jamadermatol.2021.3459. PMID: 34524406; PMCID: PMC8444064.
16. Hawkins C.M., Chewning R.H. Diagnosis and Management of Extracranial Vascular Malformations in Children: Arteriovenous Malformations, Venous Malformations, and Lymphatic Malformations. Semin Roentgenol. 2019; 54(4): 337-348. https://doi.org/10.1053/j.ro.2019.06.004. Epub 2019 Jun 26. PMID: 31706367.
17. Donyush E.K., Kondrashova Z.A., Polyaev Yu.A., Garbuzov R.V. Sirolimus for the treatment of vascular anomalies in children. Russian Journal of Pediatric Hematology and Oncology. 2020; 7(3): 22-31. (In Russ.) https://doi.org/10.21682/2311-1267-2020-7-3-22-31
Review
For citations:
Druzhinina E.S., Isaev I.V., Kostylev F.A., Narbutov A.G., Zavadenko N.N. Venous malformation as a rare cause of sciatic neuropathy. L.O. Badalyan Neurological Journal. 2023;4(3):175-180. (In Russ.) https://doi.org/10.46563/2686-8997-2023-4-3-175-180. EDN: qyuwmr