

Complexity in the diagnosis of endocrine ophthalmopathy with a carotid-cavernous fistula. A clinical case
https://doi.org/10.33791/2222-4408-2024-2-110-114
Abstract
Introduction. Clinical cases featuring a combination of endocrine ophthalmopathy with a carotid-cavernous fistula are scarce, with only four cases documented in the literature, thus posing a diagnostic challenge due to overlapping clinical symptoms. A carotid-cavernous fistula with high blood flow typically presents a clear clinical picture. However, low-flow variants are challenging to timely diagnose. A history of head trauma can be an essential diagnostic clue, as carotid-cavernous anastomosis develops in 75% of cases post-trauma and in 25% spontaneously. Bilateral carotid-cavernous fistulas are particularly rare.
Purpose: to present the clinical features and outcomes of bilateral endocrine ophthalmopathy associated with a non-traumatic carotid-cavernous fistula, based on the prospective observation of a patient treated in various city healthcare hospitals during 2021–2022.
Case description. The patient first visited the ophthalmologist in October 2021, presenting with bilateral exophthalmos, edema, and eye redness. A positive test for antibodies to thyroid-stimulating hormone and enlargement of the eye muscles, confirmed by orbital ultrasound, led to a diagnosis of “Endocrine Ophthalmopathy”. The patient’s condition was managed jointly by an endocrinologist and an ophthalmologist. Over two months, a worsening of ocular symptoms was observed, prompting a neurosurgical consultation. Cerebral angiography conducted in a hospital setting confirmed a carotid-cavernous fistula. An endovascular procedure was performed five months after the initial ocular symptoms appeared to separate the carotid-cavernous anastomosis on the right using a non-adhesive composition. Ocular symptoms resolved within 2–3 weeks post-surgery.
Conclusions. Clinical manifestations of carotid-cavernous fistulas can resemble those of endocrine ophthalmopathy. The absence of a traumatic head injury history, complaints of unilateral head noise synchronous with the pulse, and the bilateral nature of the symptoms complicated the diagnosis of this neurosurgical pathology in a patient with endocrine ophthalmopathy. It is crucial to include both conditions in the differential diagnosis and to employ a multidisciplinary approach for effective management.
Keywords
About the Authors
E. Yu. TsyplinaRussian Federation
Ekaterina Yu. Tsyplina - Student.
2, Moskovsky Trakt, Tomsk, 634050
A. A. Pochechueva
Russian Federation
Anastasia A. Pochechueva - Ophthalmologist.
22, Oktyabrsky Ave., Kemerovo, 650066
T. V. Yanchenko
Russian Federation
Tatyana V. Yanchenko - Cand. Sci. (Med.), Head of the Pediatric Surgical Department.
22, Oktyabrsky Ave., Kemerovo, 650066
G. G. Basova
Russian Federation
Galina G. Basova - Cand. Sci. (Med.), Associate Professor of the Department of Ophthalmology.
22а, Voroshilov Str., Kemerovo, 650056
References
1. Esina MV, Kuzneczova OA, Yamashkina EI, Burnaeva YV, Soldatov VN. Analysis of prevalence of endocrine ophthalmopathy in patients with autoimmune thyroid pathology. Ogarev-online. 2020;1. (In Russ.) https://journal.mrsu.ru/arts/ocenka-rasprostranennosti-endokrinnojoftalmopatii-sredi-pacientov-s-autoimmunnoj-patologiejshhitovidnoj-zhelezy
2. Nikonova LV, Tishkovskiy SV, Hadomskaya VI, Davydchyk EV, Doroshkevich IP. Endocrine ophthalmopathy: etiology, pathogenesis, clinical picture, diagnosis. Journal of the Grodno State Medical University. 2018;16(1):69–74. (In Russ.) doi: 10.25298/2221-8785-2018-16-1-69-76
3. Brovkina AF. Endocrine ophthalmopathy: reality and prospects. Ophthalmological Statements. 2012;5(2):31–34. (In Russ.)
4. Bhattarai HB, Thapaliya I, Dhungana S, Singh PB, Bhattarai M, Pokhrel B, et al. Unilateral proptosis in a patient with thyroid eye disease: A case report. SAGE Open Medical Case Reports. 2023;11. doi: 10.1177/2050313X231190669
5. Azzam DB, Cypen SG, Conger JR, Tao JP. Carotid-cavernous sinus fistula masquerading as thyroid eye disease. Cureus. 2021;13(4):e14261. doi: 10.7759/cureus.1426
6. Lacorzana J, Rocha-de-Lossada C, Ortiz-Perez S. A tricky case of unilateral orbital inflammation: carotid cavernous fistula in Graves-Basedow disease. Rom J Ophthalmol. 2021;65(2):201–203. doi: 10.22336/rjo.2021.40
7. Cossu G, Al-Taha K, Hajdu SD, Daniel RT, Messerer M. Carotidcavernous fistula after transsphenoidal surgery: A rare but challenging complication. World Neurosurg. 2020;134:221–227. doi: 10.1016/j.wneu.2019.10.194
8. Jozef Č. Carotid-cavernous fistula from the perspective of an ophthalmologist A Review. Cesk Slov Oftalmol. 2020;1(Ahead of print):1–8. doi: 10.31348/2020/8
9. Docherty G, Eslami M, Jiang K, Barton JS. Bilateral carotidcavernous sinus fistula: a case report and review of the literature. J Neurol. 2018;265(3):453–459. doi: 10.1007/s00415-017-8657-y
10. Hübner L, Struffert T, Mardin CY, Engelhorn T, Holbach L, Weller J, et al. Klinische und echographische Untersuchungsbefunde bei Patienten mit Carotis-Sinuscavernosus-Fisteln [Clinical and sonographic examination findings in patients with carotid-cavernous sinus fistulas]. Ophthalmologe. 2021;118(9):919–925. doi: 10.1007/s00347-020-01310-3
11. Sharma R, Ponder C, Kamran M, Chacko J, Kapoor N, Mylavarapu K, et al. Bilateral carotid-cavernous fistula: A diagnostic and therapeutic challenge. J Investig Med High Impact Case Rep. 2022;10:23247096221094181. doi: 10.1177/23247096221094181
12. Agrawal M, Kumari L, Vichare N, Shyamsundar K, Avasthi A, Gupta S. Carotid-cavernous fistula masquerading as thyroid associated orbitopathy: a diagnostic challenge. Rom J Ophthalmol. 2022;66(2):168–172. doi: 10.22336/rjo.2022.33
13. Urdapilleta-Contreras M, Padilla-Pérez L, Aguilar-Ruiz A, Verdugo-Unigarro A. Thyroid orbitopathy masked a carotid-cavernous fistula. Case report. Arch Soc Esp Oftalmol (Engl Ed). 2019;94(8):400–404. doi: 10.1016/j.oftal.2019.05.002
Review
For citations:
Tsyplina E.Yu., Pochechueva A.A., Yanchenko T.V., Basova G.G. Complexity in the diagnosis of endocrine ophthalmopathy with a carotid-cavernous fistula. A clinical case. The EYE GLAZ. 2024;26(2):110-114. (In Russ.) https://doi.org/10.33791/2222-4408-2024-2-110-114