Journal of Kerman University of Medical Sciences

Multiple Schwannomas Involving Unusual Communicating Loop between Ulnar and Median Nerve in Axilla: Clinical Implications

Document Type : Case Report

Authors

Dibakar Borthakur 1
Rajesh Kumar 2
Kishore Sesham 3
Harisha Kusuma 1
Rima Dada 1

1 Department of Anatomy, All India Institute of Medical Sciences, New Delhi, India

2 Department of Anatomy, All India Institute of Medical Sciences, Patna, India

3 Department of Anatomy, All India Institute of Medical Sciences, Mangalagiri, India

https://doi.org/10.34172/jkmu.2024.52
Abstract
 
 Background: Schwannoma and neurofibroma constitute benign peripheral nerve sheath tumors (BPNSTs), which may present as single or multiple lesions. Multiple schwannomas are rare in the peripheral nerves of the upper extremity. Solitary small schwannoma is usually asymptomatic, while larger and multiple schwannomas may have different symptoms due to pressure effects. Median and ulnar nerve communications are often encountered in the upper limb, making these anastomoses clinically important. However, such communications with schwannomas in the axilla are rare.
Case Report: The study protocol was designed per the prevailing guidelines of the Institute on the use of human cadavers for teaching and research. Written informed consent was obtained from the family of the body donor at the time of whole-body donation. The upper extremities, including the brachial plexuses of both sides, were carefully dissected as per the instructions in Cunningham’s Manual of Practical Anatomy15th edition.
Discussion: Unusual anastomoses in the axilla and two encapsulated, highly vascular fusiform masses were observed. Another fairly large mass was noted on the right chest wall. Histological examination confirmed the masses to be schwannomas.
Conclusion: Asymptomatic schwannomas often remain undiagnosed, and those lying in the axilla can be misdiagnosed. Awareness of such variant anatomy will enable clinicians and surgeons to plan more appropriate diagnostic and therapeutic interventions.

Keywords

Axillary mass
Median-ulnar nerve communications
Multiple schwannomas
BPNSTs
Mammae erraticae
Subjects
  1. Ünver Doğan N, Uysal II, Şeker M. The communications between the ulnar and median nerves in upper limb. Neuroanatomy. 2009;8(1):15-9.
  2. Smith JL, Siddiqui SA, Ebraheim NA. Comprehensive summary of anastomoses between the median and ulnar nerves in the forearm and hand. J Hand Microsurg. 2019;11(1):1-5. doi: 10.1055/s-0038-1672335.
  3. Kate NN, Teli CG, Gajbhiye R, Ambareesha K, Suresh M. A study to analyze the prevalence of nervous anastomosis (Martin- Gruber) in medical students. Natl J Physiol Pharm Pharmacol. 2015;5(3):1-5. doi: 10.5455/njppp.2015.5.1207201414.
  4. Albert P, Patel J, Badawy K, Weissinger W, Brenner M, Bourhill I, et al. Peripheral nerve schwannoma: a review of varying clinical presentations and imaging findings. J Foot Ankle Surg. 2017;56(3):632-7. doi: 10.1053/j.jfas.2016.12.003.
  5. Cavanagh NP, Pincott JR. Ulnar nerve tumours of the hand in childhood. J Neurol Neurosurg Psychiatry. 1977;40(8):795- 800. doi: 10.1136/jnnp.40.8.795.
  6. Artico M, Cervoni L, Wierzbicki V, D’Andrea V, Nucci F. Benign neural sheath tumours of major nerves: characteristics in 119 surgical cases. Acta Neurochir (Wien). 1997;139(12):1108- 16. doi: 10.1007/bf01410969.
  7. Tian J, Huang Q, Chen Z. Schwannoma of the long thoracic nerve in the left axilla: a case report. J Int Med Res. 2020;48(3):300060519890197. doi: 10.1177/0300060519890197.
  8. Izumi AK, Rosato FE, Wood MG. Von Recklinghausen’s disease associated with multiple neurolemomas. Arch Dermatol. 1971;104(2):172-6.
  9. Panwar P, Kumar S, Singh S, Sriharsha AS, Gupta K. Giant abdominoperineal malignant schwannoma: an unusual presentation and surgical challenge. Case Rep Urol. 2015;2015:728062. doi: 10.1155/2015/728062.
  10. Zou F, Dai M, Zhang B, Nie T. Misdiagnosis of a giant intrapelvic schwannoma: a case report. Oncol Lett. 2013;6(6):1646-8. doi: 10.3892/ol.2013.1634.
  11. Babuccu O, Kalayci M, Turhan E, Gun BD, Dursun A. Mammae erraticae: a case report and reappraisal of the related theories. Aesthetic Plast Surg. 2012;36(3):607-10. doi: 10.1007/s00266-011-9854-1.
  12. Leibovic SJ, Hastings H 2nd. Martin-Gruber revisited. J Hand Surg Am. 1992;17(1):47-53. doi: 10.1016/0363- 5023(92)90112-3.
  13. Hepburn D. The Anatomy and Comparative Anatomy of the Muscles and Nerves of the Superior and Inferior Extremities of the Anthropoid Apes [dissertation]. University of Edinburgh; 1891.
  14. Choudhary R, Agarwal D, Dixit GS, Singh B, Ghatak S. Communication between the median and musculocutaneous nerve and its clinical significance–a case report. J Anat Soc India. 2017;66:S121.
  15. Cavalheiro CS, Filho MR, Pedro G, Caetano MF, Vieira LA, Caetano EB. Clinical repercussions of Martin- Gruber anastomosis: anatomical study. Rev Bras Ortop. 2016;51(2):214-23. doi: 10.1016/j.rboe.2016.02.003.
  16. Kazakos KJ, Smyrnis A, Xarchas KC, Dimitrakopoulou A, Verettas DA. Anastomosis between the median and ulnar nerve in the forearm. An anatomic study and literature review. Acta Orthop Belg. 2005;71(1):29-35.
  17. Felippe MM, Telles FL, Soares AC, Felippe FM. Anastomosis between median nerve and ulnar nerve in the forearm. J Morphol Sci. 2017;29(1):23-6.
  18. Kara AB, Elvan Ö, Öztürk NC, Öztürk AH. Communications of the median nerve in foetuses. Folia Morphol (Warsz). 2018;77(3):441-6. doi: 10.5603/FM.a2017.0107.
  19. Pertea M, Filip A, Huzum B, Lunca S, Carp C, Mitrea M, et al. Schwannoma of the upper limb: retrospective study of a rare tumor with uncommon locations. Diagnostics (Basel). 2022;12(6):1319. doi: 10.3390/diagnostics12061319.
  20. Ardeleanu V, Pirici D, Sava A, Folescu R, Motoc AG. Cystic schwannoma of the distal forearm. Case presentation. Rom J Morphol Embryol. 2020;61(3):911-6. doi: 10.47162/ rjme.61.3.30.
  21. Fodor L, Samuila S, Bodog F. A rare case of schwannomatosis of the extremities. J Int Med Res. 2020;48(9):300060520952278. doi: 10.1177/0300060520952278.
  22. Hakan T, Kılıç Y, Çelikoğlu E, Ekemen S. An Unusual Schwannoma in the Proximal Forearm: A Case Report. Cureus. 2019;11(11):e6231. doi: 10.7759/cureus.6231.
  23. Xarchas KC, Kyriakopoulos G, Vlachou M, Manthas S. Median nerve schwannoma of the hand and wrist: 3 cases. Open Orthop J. 2019;13(1):290-4. doi: 10.2174/1874325001913010290.
  24. Vučemilo L, Lajtman Z, Mihalj J, Plašćak J, Mahović Lakušić D, Mužinić D. Brachial plexus schwannoma - case report and literature review. Acta Clin Croat. 2018;57(2):366-71. doi: 10.20471/acc.2018.57.02.19.
  25. Tabrizi A, Afshar A, Mohebbi I, Pourjabali M, Taleb H. Schwannoma and neurofibroma, originating from the ulnar nerve in neurofibromatosis: a case report and review of the literature. Surg J (N Y). 2020;6(3):e139-44. doi: 10.1055/s- 0040-1712536.
  26. Nishida M, Kawano Y, Yuge A, Nasu K, Matsumoto H, Narahara H. Three cases of immature teratoma diagnosed after laparoscopic operation. Clin Med Insights Case Rep. 2014;7:91-4. doi: 10.4137/CCRep.S17455.
  27. Dusad T, Meena DS, Saini N, Sharma Y, Khurana D. Schwannoma of the median nerve at mid forearm level. J Orthop Case Rep. 2016;6(2):66-8. doi: 10.13107/jocr.2250- 0685.438.
  28. Battaglia PJ, Carbone-Hobbs V, Guebert GM, Mackinnon SE, Kettner NW. High-resolution ultrasonography and shear-wave sonoelastography of a cystic radial nerve Schwannoma. J Ultrasound. 2017;20(3):261-6. doi: 10.1007/s40477-017- 0254-5.
  29. Padasali PS, Shankaregowda VS, Kshirsagar SD. Median nerve schwannoma: a case and review of literature. Asian J Neurosurg. 2015;10(3):212-5. doi: 10.4103/1793- 5482.161178.
  30. Rammohan R, Gupta P, Maini L, Gautam VK. Neurilemmoma of median nerve. J Clin Orthop Trauma. 2014;5(1):33-7. doi: 10.1016/j.jcot.2014.02.002.
  31. Anghel A, Tudose I, Terzea D, Răducu L, Sinescu RD. Unusual median nerve schwannoma: a case presentation. Rom J Morphol Embryol. 2014;55(1):159-64.
  32. Kütahya H, Güleç A, Güzel Y, Kacira B, Toker S. Schwannoma of the median nerve at the wrist and palmar regions of the hand: a rare case report. Case Rep Orthop. 2013;2013:950106. doi: 10.1155/2013/950106.
  33. Tang CY, Fung B, Fok M, Zhu J. Schwannoma in the upper limbs. Biomed Res Int. 2013;2013:167196. doi: 10.1155/2013/167196.
  34. Kumar A, Akhtar S. Schwannoma of brachial plexus. Indian J Surg. 2011;73(1):80-1. doi: 10.1007/s12262-010-0141-1.
  35. Kwon JY, Chung KW, Park EK, Park SW, Choi BO. Charcot- Marie-Tooth 1A concurrent with schwannomas of the spinal cord and median nerve. J Korean Med Sci. 2009;24(4):763-6. doi: 10.3346/jkms.2009.24.4.763.
  36. Aydin MD, Kotan D, Keles M. Acute median nerve palsy due to hemorrhaged schwannoma: case report. J Brachial Plex Peripher Nerve Inj. 2007;2:19. doi: 10.1186/1749-7221-2-19.
  37. Chen F, Miyahara R, Matsunaga Y, Koyama T. Schwannoma of the brachial plexus presenting as an enlarging cystic mass: report of a case. Ann Thorac Cardiovasc Surg. 2008;14(5):311-3.
  38. Knight DM, Birch R, Pringle J. Benign solitary schwannomas: a review of 234 cases. J Bone Joint Surg Br. 2007;89(3):382-7. doi: 10.1302/0301-620x.89b3.18123.
  39. Jiang S, Shen H, Lu H. Multiple schwannomas of the digital nerves and common palmar digital nerves: an unusual case report of multiple schwannomas in one hand. Medicine (Baltimore). 2019;98(10):e14605. doi: 10.1097/ md.0000000000014605.
  40. Kühn JP, Wagner M, Bozzato A, Linxweiler M. Multiple schwannomas of the facial nerve mimicking cervical lymphoma: a case report. J Med Case Rep. 2021;15(1):436. doi: 10.1186/s13256-021-03006-x.
  41. Shariatzadeh H, Amiri S, Joudi S, Bahrabadi M. Multiple schwannomas in sciatic nerve: a rare case report. Arch Bone Jt Surg. 2021;9(5):601-4. doi: 10.22038/abjs.2021.53165.2638.
  42. Gosk J, Gutkowska O, Kuliński S, Urban M, Hałoń A. Multiple schwannomas of the digital nerves and superficial radial nerve: two unusual cases of segmental schwannomatosis. Folia Neuropathol. 2015;53(2):158-67. doi: 10.5114/ fn.2015.52413.
  43. Huang JH, Samadani U, Zager EL. Brachial plexus region tumors: a review of their history, classification, surgical management, and outcomes. Neurosurg Q. 2003;13(3):151-61.
  44. Biswas D, Marnane CN, Mal R, Baldwin D. Extracranial head and neck schwannomas--a 10-year review. Auris Nasus Larynx. 2007;34(3):353-9. doi: 10.1016/j.anl.2007.01.006.
  45. Van Herendael B, Heyman S, De Schepper AM, Gielen J, Parizel PM. Schwannoma of left ulnar nerve. JBR-BTR. 2006;89(3):156-7.
  46. Visser LH. High-resolution sonography of the superficial radial nerve with two case reports. Muscle Nerve. 2009;39(3):392-5. doi: 10.1002/mus.21246.
  47. Lekanne Deprez RH, Bianchi AB, Groen NA, Seizinger BR, Hagemeijer A, van Drunen E, et al. Frequent NF2 gene transcript mutations in sporadic meningiomas and vestibular schwannomas. Am J Hum Genet. 1994;54(6):1022-9.
  48. Evans DG, Bowers NL, Tobi S, Hartley C, Wallace AJ, King AT, et al. Schwannomatosis: a genetic and epidemiological study. J Neurol Neurosurg Psychiatry. 2018;89(11):1215-9. doi: 10.1136/jnnp-2018-318538.
  49. Smith MJ, Isidor B, Beetz C, Williams SG, Bhaskar SS, Richer W, et al. Mutations in LZTR1 add to the complex heterogeneity of schwannomatosis. Neurology. 2015;84(2):141-7. doi: 10.1212/wnl.0000000000001129.
  50. Pathmanaban ON, Sadler KV, Kamaly-Asl ID, King AT, Rutherford SA, Hammerbeck-Ward C, et al. Association of genetic predisposition with solitary schwannoma or meningioma in children and young adults. JAMA Neurol. 2017;74(9):1123-9. doi: 10.1001/jamaneurol.2017.1406.

 

Journal of Kerman University of Medical Sciences
Volume 31, Issue 6
November and December 2024
Pages 338-343

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