Trigeminal trophic syndrome
DOI: 10.32364/2587-6821-2020-4-9-566-572
V.V. Belopasov
Astrakhan State Medical University, Astrakhan, Russian Federation
Trigeminal trophic syndrome is a rare nosological pathology form of the face. Based on the literature data and 11 own observations, the article discusses the pathogenesis, diagnosis and treatment of skin sensations, ulcerative and other cosmetic defects that occur after injury of the peripheral branches, Gasser's node, root, and the upper sensitive trigeminal nerve nucleus in the dorsolateral part of the pons Varolii upper section in the brain stem. The clinical disease manifestations are described in detail with the characteristics of the lesions, their localization, course dynamics, and mental status patterns of patients. The article presents a detailed list of pathological conditions with which trigeminal trophic syndrome should be differentiated (malignant epithelioma, mycoses, focal fibrosing alopecia, leprosy, syphilis, diabetes mellitus, cutaneous manifestations in diseases of internal organs and connective tissue (scleroderma, temporal arteritis, Wegen er's granulomatosis), central and peripheral nervous system, substance abuse, psychosomatic disorders, somatopsychosis, autoaggression, Munchausen syndrome, etc.).
Keywords: skin ulcers, neurotrophic disorders, trigeminal trophic syndrome, deafferentation, iatrogenic complications.
For citation: Belopasov V.V. Trigeminal trophic syndrome. Russian Medical Inquiry. 2020;4(9):566–572. DOI: 10.32364/2587-6821-2020-4-9-566-572.
About the author:
Vladimir V. Belopasov — Dr. of Sci. (Med.), Professor, Head of the Department of Neurology and Neurosurgery with Postgraduate Education course, Astrakhan State Medical University: 121, Bakinskaya str., Astrakhan, 414000, Russian Federation.
Contact information: Vladimir V. Belopasov, e-mail: belopasov@yandex.ru. Financial Disclosure: the author has not a financial or property interest in any material or method mentioned. There is no conflict of interests. Received 06.10.2020, revised 20.10.2020, accepted 03.11.2020.
References
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2. Grigorieva T.A. About the cause of trophic disorders in sensitive areas of the body. Reports of the USSR Academy of Sciences (new series). 1951;78(2):387–390 (in Russ.).
3. Speransky A.D. Nervous trophism in the theory and practice of medicine. M.: VIEM; 1936 (in Russ.).
4. Belopasov V.V. Leprosy. In the book: Chronic neuroinfections. M.: GEOTAR-Media; 2017 (in Russ.).
5. Wallenberg A. Klinishe Beitrage zur Diagnostik acuter Herderkrankungen des Verlagten Marks und der Bruke. Dtsch Z Nervenheilkd. 1901;19:227–231.
6. Loveman A.B. An unusual dermatosis following section of the fifth cranial nerve. Arch Dermatol Syph. 1933;28:369–375.
7. Fischer A.A., Adelson D.M., Garcia C. Cervical trophic syndrome: a distinct clinical entity? Cutis. 2014;93(6):E6–E7.
8. Bradburn K.H., Elston D., Murphey A.W., Patel K.G. Trigeminal Trophic Syndrome — A Unique Clinical Presentation of a Rare Condition. Ear Nose Throat J. 2019;98(10):606–608. DOI: 10.1177/0145561319850818.
9. Khan A.U., Khachemoune A. Trigeminal trophic syndrome: an updated review. Int J Dermatol. 2019;58(5):530–537. DOI: 10.1111/ijd.14098.
10. Grachev Yu.V. Trigeminal prosopalgia (pathogenesis, semiotics, diagnostic methods, treatment): dis. … Dr. med. sci. M.; 1996 (in Russ.).
11. Karlov V.A. Facial neurology. M.: Medicine; 1991 (in Russ.).
12. Kentley J., Marshall C., Gkini М.-A. et al. Atypical Trigeminal Trophic Syndrome: An Unusual Cause of Facial Ulceration. Acta Derm. Venereol. 2017;97:971–972. DOI: 10.2340/0001-5555-2675.
13. Rashid R.M., Khachemoune A. Trigeminal trophic syndrome. J Eur Acad Dermatol Venereol. 2007;21(6):725–731. DOI: 10.1111/j.1468-3083.2007.02250.x.
14. Golden E., Robertson C.E., Moossy J.J. et al. Trigeminal trophic syndrome: A rare cause of chronic facial pain and skin ulcers. Cephalalgia. 2015;35(7):636–636. DOI: 10.1177/0333102-41-4547140.
15. Di Luca A., Ralli M., Hemied S. et al. An atypical case of trigeminal trophic syndrome: A legal medicine perspective in medical responsibility. SAGE Open Med Case Rep. 2017;5:2050313X17723548. DOI: 10.1177/2050313X17723548.
16. Setyadi H.G., Cohen P.R., Schulze K.E. et al. Trigeminal trophic syndrome. South Med J. 2007;100(1):43–48. DOI: 10.1097/01.smj.0000253020.74133.7e.
17. Morales-Raya C., García-González E., Maroñas-Jiménez L. Trigeminal trophic syndrome: An unusual cause of nasal ulceration. Acta Otorinolaringol. Esp. 2017;68(3):188–189. DOI: 10.1016/j.otorri.2016.02.009.
18. Farahani R.M., Marsee D.K., Baden L.R. et al. Trigeminal trophic syndrome with features of oral CMV disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008;106(3):e15–e18. DOI: 10.1016/j.tripleo.2008. 05.002.
19. Palanisamy A., Rajappavu S.D., Kothandapany S. Trigeminal trophic syndrome. An Bras Dermatol. 2017;92(4):593–594. DOI: 10.1590/abd1806-4841.20175484.
20. Pedicelli C., Paradisi A., Fazio M. et al. Trigeminal neurotrophic ulceration in Wallenberg’s syndrome. Int J Dermatol. 2009;48(4):443–445. DOI: 10.1111/j.1365-4632.2009.03920.x.
21. Dolohanty L.B., Richardson S.J., Herrmann D.N. et al. Trigeminal trophic syndrome with histopathologic correlation. Cutis. 2015;95(3):E22–E25.
22. Bove D., Lupoli A., Caccavale S. et al. Dermatological and immunological conditions due to nerve lesions. Funct Neurol. 2013;28(2):83–91. DOI: 10.11138/FNeur/2013.28.2.083.
23. Kumar P., Thomas J. Trigeminal Trophic Syndrome. Ind J Dermatol. 2014;59(1):75–76. DOI: 10.4103/0019-5154.123506.
24. Gupta M.A., Vujcic B., Gupta A.K. Dissociation and conversion symptoms in dermatology. Clin Dermatol. 2017;35(3):267–272. DOI: 10.1016/j.clindermatol.2017.01.003.
25. Lvov A.N. Dermatoses comorbid with mental disorders: classification, clinic, therapy and prevention: dis. … Dr. med. sci. M.; 2006 (in Russ.).
26. Dorozhenok I.Yu. Nosogenic disorders in skin diseases. In: Lectures on psychosomatics. A.B. Smulevich, ed. M.: Medical Information Agency; 2014 (in Russ.).
27. Shumway N.K., Cole E., Fernandez K.H. Neurocutaneous disease: Neurocutaneous dysesthesias. J Am Acad Dermatol. 2016;74(2):215–228. DOI: 10.1016/j.jaad.2015.04.059.
28. Uchiyama A., Motegi S.I., Fujiwara C. et al. Trigeminal trophic syndrome: Analysis of the number of peripheral nerve fibres and blood vessels in the lesional skin. Australas J. Dermatol. 2018;59(1):59–61. DOI: 10.1111/ajd.12647.
29. Gan E.Y., Tey H.L. The ‘Phantom of the Opera’ ulcer. Clin Exp Dermatol. 2012;37(5):581–822. DOI: 10.1111/j.1365-2230.2011.04222.x.
30. McVeigh K.A., Adams M., Harrad R., Ford R. Periocular manifestations of trigeminal trophic syndrome: A case series and literature review. Orbit. 2018;37(1):32–35. DOI: 10.1080/01676830.2017.1353117.
31. Zotti F., Capocasale G., Lonardi F. et al. Trigeminal trophic syndrome: Strange evolution of maxillofacial surgery. EXCLI J. 2019;18:931–935. DOI: 10.17179/excli2019-1846.
32. Smulevich A.B., Romanova D.V., Lvov A.N. and other Dermatozoal delusions and associated disorders. M.: GEOTAR-Media; 2015 (in Russ.).
33. Bhatti A.F., Soggiu D., Orlando A. Trigeminal trophic syndrome: diagnosis and management difficulties. Plast Reconstr Surg. 2008;121(1):1e–3e. DOI: 10.1097/01.prs.0000293759.03 833.88.
34. Nemet A.Y., Sterenberg I., Rosenman Y. Full thickness eyelid defect in a trigeminal trophic syndrome. Arch Ophthalmol. 2007;125(10):1438–1439. DOI: 10.1001/archopht.12 5.10.1438.
35. Wu M.Y., Liu R.F., Chang Y.Y. Image Gallery: Trigeminal trophic syndrome: nonhealing facial ulcer with corneal ulceration. Br J Dermatol. 2019;181(1): e2. DOI: 10.1111/bjd.17785.
36. Tabashnikova T.V., Serova N.K., Shimanskiy V.N. Trophic keratopathy treatment and lagophthalmos correction in neurosurgical patients. Ophthalmology journal. 2013;6(3):9–15 (in Russ.). DOI: 10.17816/OV201339-15
37. Dua H.S., Said D.G., Messmer E.M. et al. Neurotrophic keratopathy. Prog Retin Eye Res. 2018;66:107–131. DOI: 10.1016/j.preteyeres. 2018.04.003.
38. Nair M., Ajila V., Hegde S. et al. Clinical and radiographic features of parry-romberg syndrome. J Istanb Univ Fac Dent. 2017;51(3):45–49. DOI: 10.17096/jiufd.72612.
39. Sadeghi P., Papay F.A., Vidimos A.T. Trigeminal trophic syndrome-report of four cases and review of the literature. Dermatol Surg. 2004;30(5):807–812. DOI: 10.1111/j.1524-4725.2004.30220.x.
40. Shchedrenok V.V., Topolskaya N.V., Zakhmatova T.V. and others. Facial and headache. Clinical and radiation diagnostics and surgical treatment. SPb.: Publishing house of the Leningrad Regional Institute of Education Development, 2013 (in Russ.).
41. Fernández-Bernáldez A., Rodríguez-Jiménez P., Herrero-Moyano M., Sánchez-Pérez J. Trigeminal Trophic Syndrome Secondary to Meningioma Recurrence. Actas Dermosifiliogr. 2020;111(2):171–173. DOI: 10.1016/j.ad.2018.08.013.
42. Carr P., Martin S., Young J., Chiota-McCollum N. Trigeminal trophic syndrome: A possible dermatologic manifestation of stroke deficits. Neurol Clin Pract. 2020;10(4): e27–e29. DOI: 10.1212/CPJ.0000000000000711.
43. Elhennawy K., Reda S., Finke C. et al. Oral manifestations, dental management, and a rare homozygous mutation of the PRDM12 gene in a boy with hereditary sensory and autonomic neuropathy type VIII: a case report and review of the literature. J Med Case Rep. 2017;11(1):233. DOI: 10.1186/s13256-017-1387-z.
44. Otsuka T., Iwata H., Kosumi H. et al. Refractory oral ulcers in eosinophilic granulomatosis with polyangiitis. J Dermatol. 2019;46(10): e377–e378. DOI: 10.1111/1346-81-38. 14897.
45. Hachisuka J., Chiang M.C., Ross S.E. Itch and neuropathic itch. Pain. 2018;159(3):603–609. DOI: 10.1097/j.pain.0000000000001141.
46. Sousa Filho D., Kanomata E.Y., Feldman R.J., Maluf Neto A. Munchausen syndrome and Munchausen syndrome by proxy: a narrative review. Einstein (Sao Paulo). 2017;15(4):516–521. DOI: 10.1590/S1679-45082017MD3746.
47. Smulevich A.B., Lvov A.N., Ivanov O.L. Pathomimies: the psychopathology of autoaggression in dermatological practice. M.: Medical Information Agency; 2012 (in Russ.).
48. Sallabanda K., Sallabanda M., Barrientos H.D. et al. Trigeminal Trophic Syndrome Secondary to Refractory Trigeminal Neuralgia Treated with CyberKnife® Radiosurgery. Cureus. 2020;12(4):e7670. DOI: 10.7759/cureus.7670.
49. Matsuda K.M., Tanaka-Mizutsugu H., Kishi Y., Hino H., Kagami S. A case of trigeminal trophic syndrome responding to mirogabalin. Eur J Dermatol. 2020. DOI: 10.1684/ejd.2020.3746. Online ahead of print.
50. Hufschmidt K., Fernandez J., Balaguer T. et al. Traitement de l’ulcération neurotrophique après alcoolisation du ganglion de Gasser dans l’atteinte du nerf trijumeau: cas clinique et revue de la littérature. Ann Chir Plast Esthet. 2017;62(1):79–86. DOI:10.1016/j.anplas.2016.03.004.
2. Grigorieva T.A. About the cause of trophic disorders in sensitive areas of the body. Reports of the USSR Academy of Sciences (new series). 1951;78(2):387–390 (in Russ.).
3. Speransky A.D. Nervous trophism in the theory and practice of medicine. M.: VIEM; 1936 (in Russ.).
4. Belopasov V.V. Leprosy. In the book: Chronic neuroinfections. M.: GEOTAR-Media; 2017 (in Russ.).
5. Wallenberg A. Klinishe Beitrage zur Diagnostik acuter Herderkrankungen des Verlagten Marks und der Bruke. Dtsch Z Nervenheilkd. 1901;19:227–231.
6. Loveman A.B. An unusual dermatosis following section of the fifth cranial nerve. Arch Dermatol Syph. 1933;28:369–375.
7. Fischer A.A., Adelson D.M., Garcia C. Cervical trophic syndrome: a distinct clinical entity? Cutis. 2014;93(6):E6–E7.
8. Bradburn K.H., Elston D., Murphey A.W., Patel K.G. Trigeminal Trophic Syndrome — A Unique Clinical Presentation of a Rare Condition. Ear Nose Throat J. 2019;98(10):606–608. DOI: 10.1177/0145561319850818.
9. Khan A.U., Khachemoune A. Trigeminal trophic syndrome: an updated review. Int J Dermatol. 2019;58(5):530–537. DOI: 10.1111/ijd.14098.
10. Grachev Yu.V. Trigeminal prosopalgia (pathogenesis, semiotics, diagnostic methods, treatment): dis. … Dr. med. sci. M.; 1996 (in Russ.).
11. Karlov V.A. Facial neurology. M.: Medicine; 1991 (in Russ.).
12. Kentley J., Marshall C., Gkini М.-A. et al. Atypical Trigeminal Trophic Syndrome: An Unusual Cause of Facial Ulceration. Acta Derm. Venereol. 2017;97:971–972. DOI: 10.2340/0001-5555-2675.
13. Rashid R.M., Khachemoune A. Trigeminal trophic syndrome. J Eur Acad Dermatol Venereol. 2007;21(6):725–731. DOI: 10.1111/j.1468-3083.2007.02250.x.
14. Golden E., Robertson C.E., Moossy J.J. et al. Trigeminal trophic syndrome: A rare cause of chronic facial pain and skin ulcers. Cephalalgia. 2015;35(7):636–636. DOI: 10.1177/0333102-41-4547140.
15. Di Luca A., Ralli M., Hemied S. et al. An atypical case of trigeminal trophic syndrome: A legal medicine perspective in medical responsibility. SAGE Open Med Case Rep. 2017;5:2050313X17723548. DOI: 10.1177/2050313X17723548.
16. Setyadi H.G., Cohen P.R., Schulze K.E. et al. Trigeminal trophic syndrome. South Med J. 2007;100(1):43–48. DOI: 10.1097/01.smj.0000253020.74133.7e.
17. Morales-Raya C., García-González E., Maroñas-Jiménez L. Trigeminal trophic syndrome: An unusual cause of nasal ulceration. Acta Otorinolaringol. Esp. 2017;68(3):188–189. DOI: 10.1016/j.otorri.2016.02.009.
18. Farahani R.M., Marsee D.K., Baden L.R. et al. Trigeminal trophic syndrome with features of oral CMV disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008;106(3):e15–e18. DOI: 10.1016/j.tripleo.2008. 05.002.
19. Palanisamy A., Rajappavu S.D., Kothandapany S. Trigeminal trophic syndrome. An Bras Dermatol. 2017;92(4):593–594. DOI: 10.1590/abd1806-4841.20175484.
20. Pedicelli C., Paradisi A., Fazio M. et al. Trigeminal neurotrophic ulceration in Wallenberg’s syndrome. Int J Dermatol. 2009;48(4):443–445. DOI: 10.1111/j.1365-4632.2009.03920.x.
21. Dolohanty L.B., Richardson S.J., Herrmann D.N. et al. Trigeminal trophic syndrome with histopathologic correlation. Cutis. 2015;95(3):E22–E25.
22. Bove D., Lupoli A., Caccavale S. et al. Dermatological and immunological conditions due to nerve lesions. Funct Neurol. 2013;28(2):83–91. DOI: 10.11138/FNeur/2013.28.2.083.
23. Kumar P., Thomas J. Trigeminal Trophic Syndrome. Ind J Dermatol. 2014;59(1):75–76. DOI: 10.4103/0019-5154.123506.
24. Gupta M.A., Vujcic B., Gupta A.K. Dissociation and conversion symptoms in dermatology. Clin Dermatol. 2017;35(3):267–272. DOI: 10.1016/j.clindermatol.2017.01.003.
25. Lvov A.N. Dermatoses comorbid with mental disorders: classification, clinic, therapy and prevention: dis. … Dr. med. sci. M.; 2006 (in Russ.).
26. Dorozhenok I.Yu. Nosogenic disorders in skin diseases. In: Lectures on psychosomatics. A.B. Smulevich, ed. M.: Medical Information Agency; 2014 (in Russ.).
27. Shumway N.K., Cole E., Fernandez K.H. Neurocutaneous disease: Neurocutaneous dysesthesias. J Am Acad Dermatol. 2016;74(2):215–228. DOI: 10.1016/j.jaad.2015.04.059.
28. Uchiyama A., Motegi S.I., Fujiwara C. et al. Trigeminal trophic syndrome: Analysis of the number of peripheral nerve fibres and blood vessels in the lesional skin. Australas J. Dermatol. 2018;59(1):59–61. DOI: 10.1111/ajd.12647.
29. Gan E.Y., Tey H.L. The ‘Phantom of the Opera’ ulcer. Clin Exp Dermatol. 2012;37(5):581–822. DOI: 10.1111/j.1365-2230.2011.04222.x.
30. McVeigh K.A., Adams M., Harrad R., Ford R. Periocular manifestations of trigeminal trophic syndrome: A case series and literature review. Orbit. 2018;37(1):32–35. DOI: 10.1080/01676830.2017.1353117.
31. Zotti F., Capocasale G., Lonardi F. et al. Trigeminal trophic syndrome: Strange evolution of maxillofacial surgery. EXCLI J. 2019;18:931–935. DOI: 10.17179/excli2019-1846.
32. Smulevich A.B., Romanova D.V., Lvov A.N. and other Dermatozoal delusions and associated disorders. M.: GEOTAR-Media; 2015 (in Russ.).
33. Bhatti A.F., Soggiu D., Orlando A. Trigeminal trophic syndrome: diagnosis and management difficulties. Plast Reconstr Surg. 2008;121(1):1e–3e. DOI: 10.1097/01.prs.0000293759.03 833.88.
34. Nemet A.Y., Sterenberg I., Rosenman Y. Full thickness eyelid defect in a trigeminal trophic syndrome. Arch Ophthalmol. 2007;125(10):1438–1439. DOI: 10.1001/archopht.12 5.10.1438.
35. Wu M.Y., Liu R.F., Chang Y.Y. Image Gallery: Trigeminal trophic syndrome: nonhealing facial ulcer with corneal ulceration. Br J Dermatol. 2019;181(1): e2. DOI: 10.1111/bjd.17785.
36. Tabashnikova T.V., Serova N.K., Shimanskiy V.N. Trophic keratopathy treatment and lagophthalmos correction in neurosurgical patients. Ophthalmology journal. 2013;6(3):9–15 (in Russ.). DOI: 10.17816/OV201339-15
37. Dua H.S., Said D.G., Messmer E.M. et al. Neurotrophic keratopathy. Prog Retin Eye Res. 2018;66:107–131. DOI: 10.1016/j.preteyeres. 2018.04.003.
38. Nair M., Ajila V., Hegde S. et al. Clinical and radiographic features of parry-romberg syndrome. J Istanb Univ Fac Dent. 2017;51(3):45–49. DOI: 10.17096/jiufd.72612.
39. Sadeghi P., Papay F.A., Vidimos A.T. Trigeminal trophic syndrome-report of four cases and review of the literature. Dermatol Surg. 2004;30(5):807–812. DOI: 10.1111/j.1524-4725.2004.30220.x.
40. Shchedrenok V.V., Topolskaya N.V., Zakhmatova T.V. and others. Facial and headache. Clinical and radiation diagnostics and surgical treatment. SPb.: Publishing house of the Leningrad Regional Institute of Education Development, 2013 (in Russ.).
41. Fernández-Bernáldez A., Rodríguez-Jiménez P., Herrero-Moyano M., Sánchez-Pérez J. Trigeminal Trophic Syndrome Secondary to Meningioma Recurrence. Actas Dermosifiliogr. 2020;111(2):171–173. DOI: 10.1016/j.ad.2018.08.013.
42. Carr P., Martin S., Young J., Chiota-McCollum N. Trigeminal trophic syndrome: A possible dermatologic manifestation of stroke deficits. Neurol Clin Pract. 2020;10(4): e27–e29. DOI: 10.1212/CPJ.0000000000000711.
43. Elhennawy K., Reda S., Finke C. et al. Oral manifestations, dental management, and a rare homozygous mutation of the PRDM12 gene in a boy with hereditary sensory and autonomic neuropathy type VIII: a case report and review of the literature. J Med Case Rep. 2017;11(1):233. DOI: 10.1186/s13256-017-1387-z.
44. Otsuka T., Iwata H., Kosumi H. et al. Refractory oral ulcers in eosinophilic granulomatosis with polyangiitis. J Dermatol. 2019;46(10): e377–e378. DOI: 10.1111/1346-81-38. 14897.
45. Hachisuka J., Chiang M.C., Ross S.E. Itch and neuropathic itch. Pain. 2018;159(3):603–609. DOI: 10.1097/j.pain.0000000000001141.
46. Sousa Filho D., Kanomata E.Y., Feldman R.J., Maluf Neto A. Munchausen syndrome and Munchausen syndrome by proxy: a narrative review. Einstein (Sao Paulo). 2017;15(4):516–521. DOI: 10.1590/S1679-45082017MD3746.
47. Smulevich A.B., Lvov A.N., Ivanov O.L. Pathomimies: the psychopathology of autoaggression in dermatological practice. M.: Medical Information Agency; 2012 (in Russ.).
48. Sallabanda K., Sallabanda M., Barrientos H.D. et al. Trigeminal Trophic Syndrome Secondary to Refractory Trigeminal Neuralgia Treated with CyberKnife® Radiosurgery. Cureus. 2020;12(4):e7670. DOI: 10.7759/cureus.7670.
49. Matsuda K.M., Tanaka-Mizutsugu H., Kishi Y., Hino H., Kagami S. A case of trigeminal trophic syndrome responding to mirogabalin. Eur J Dermatol. 2020. DOI: 10.1684/ejd.2020.3746. Online ahead of print.
50. Hufschmidt K., Fernandez J., Balaguer T. et al. Traitement de l’ulcération neurotrophique après alcoolisation du ganglion de Gasser dans l’atteinte du nerf trijumeau: cas clinique et revue de la littérature. Ann Chir Plast Esthet. 2017;62(1):79–86. DOI:10.1016/j.anplas.2016.03.004.
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