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Acute bilateral blindness in a young Covid-19 patient with rhino-orbito-cerebral mucormycosis
Journal of Ophthalmic Inflammation and Infection Pub Date : 2021-10-18 , DOI: 10.1186/s12348-021-00272-0
Ines Malek 1 , Jihene Sayadi 1 , Rim Lahiani 2 , Miriam Boumediene 3 , Memia Ben Salah 2 , Myriam Jrad 3 , Moncef Khairallah 4 , Leila Nacef 1
Affiliation  

A diabetic 20-year-old male patient, not previously vaccinated against SARS-CoV-2, was referred to our emergency department for rapid bilateral visual loss with left periorbital pain, proptosis, palpebral edema and swelling.

He had been admitted one week before in a primary Covid-19 center for COVID-19 respiratory distress syndrome and treated with corticosteroids.

Upon ophthalmic examination, both eyes had a fixed dilated pupil with no light perception. The left eye showed features of orbital cellulitis (Fig. 1) with complete ophthalmoplegia (Fig. 2). There was a mild proptosis and limited abduction in the right eye (Fig. 2), and fundus examination showed retinal whitening with a cherry red spot and segmental blood flow consistent with central retinal artery occlusion (CRAO) (Fig. 3). Swept source OCT showed in the right eye hyperreflectivity of inner retinal layers corresponding to ischemic edema (Fig. 3).

Fig. 1
figure1

A 20-year old COVID-19-affected patient presented with rhino-orbitocerebral mucormycosis. (A) Complete blepharoptosis and inflammatory signs are seen on the left side. There is a mild proptosis on the right side. (B) Note the conjunctival injection, the chemosis and the corneal oedema. (C) Coronal and (D, E) Axial contrast-enhanced brain CT scan show thickening with infiltration of left hemiface fat planes (white stars); left pre-septal collection (arrow) with exophthalmos and lengthening of the antero-posterior axis of the eyeball (large arrow). This collection reaches the cellulo-fatty tissues next to the left maxillary sinus (curved arrow), the latter is the site of a partial liquid filling. The left cavernous sinus shows signs of thrombosis (black arrow). (F) axial T2 weighted image MRI showing T2 hypersignals (large arrow) in areas of the frontal lobes confirming endocranial extension. (G) MR angiography shows left carotid artery thrombosis (arrow). (H) 3D T1 weighted sequence with contrast shows left cavernous sinus thrombosis (curved arrow)

Full size image
Fig. 2
figure2

Clinical photographies showing complete left ophthalmoplegia and limited right eye abduction

Full size image
Fig. 3
figure3

(A) Fundus photography of the right eye showing retinal whitening with a cherry red spot and segmental blood flow consistent with central retinal artery occlusion. (B) Swept source OCT scan showing hyperreflectivity of inner retinal layers

Full size image

Brain CT complemented with cerebral MRI disclosed endocranial extension of the disease and thrombosis of both left cavernous sinus and left internal carotid artery (Fig. 1).

The patient underwent emergency endoscopic sinus examination and removal of a blackish necrotic tissues from paranasal sinuses. Histopathological examination confirmed the diagnosis of mucormycosis.

The patient was started on intra-venous liposomal amphotericin B and clavulanic-acid-amoxicillin. Dexamethasone was discontinued. Debridement of the involved sinuses and adjacent structures was attempted together with diluted amphotericin B irrigation. Orbital exenteration was discussed but it was not retained due to poor prognosis and lack of the patient’s consent.

Over the following days, the patient’s ocular and general conditions worsened due to bilateral extensive eyelid and facial necrosis with purulent melting of the left eyeball and central nervous system involvement (Fig. 4). He passed away at day 30.

Fig. 4
figure4

Clinical photographies 12 days after initial presentation showing bilateral extensive eyelid necrosis with purulent melting of the left eyeball

Full size image

With the onset of COVID-19 pandemic, clinicians have seen a sudden surge of cases of mucormycosis. Although all ocular structures may be involved in patients with COVID-19 infection, orbital mucormycosis seems to be the most aggressive and so far fatal complication [1,2,3].

To the best of our knowledge very few cases of severe bilateral blindness in Covid-19 patient have been reported [2,3,4,5,6]. They were related either to orbital location of mucormycosis [2, 3] or to neurological complications including bilateral optic neuritis [4] and occipital ischemic stroke [5, 6].

The presentation of our patient suggests a left cavernous sinus and internal carotid thrombosis together with a right CRAO. From the ophthalmic and other orbital arteries, fungal infection advanced into the left cavernous sinus, ipsilateral carotid artery and right central retinal artery. This resulted in acute onset of bilateral vision loss and left ophthalmoplegia.

Successful treatment of ROCM highly depends on the early diagnosis of the infection, the control of the underlying predisposing factors and aggressive surgery [7].

Hence, Ophthalmologists should have a high index of suspicion for mucormycosis development in diabetic patients with COVID-19 illness, treated with corticosteroids. Vaccination against SARS-COv2 seems to be the best treatment to prevent COVID-19 and related blinding and fatal diseases.

For data, please refer to corresponding author.

ROCM:

Rhino-Orbito-Cerebral Mucormycosis

CRAO:

Central Retinal Artery Occlusion

  1. 1.

    Sen, M., Honavar, S. G., Sharma, N., & Sachdev, M. S. (2021). COVID-19 and eye: a review of ophthalmic manifestations of COVID-19. Indian J Ophthalmol, 69(3), 488–509. https://doi.org/10.4103/ijo. IJO_297_21

  2. 2.

    Veisi, A., Bagheri, A., Eshaghi, M., Rikhtehgar, M. H., Rezaei Kanavi, M., & Farjad, R. (2021). Rhino-orbital mucormycosis during steroid therapy in COVID-19 patients: a case report. European journal of ophthalmology, 11206721211009450. Advance online publication. https://doi.org/10.1177/11206721211009450, 112067212110094

  3. 3.

    Eswaran, S., Balan, S. K., & Saravanam, P. K. (2021). Acute fulminant Mucormycosis triggered by Covid 19 infection in a young patient. Indian journal of otolaryngology and head and neck surgery : official publication of the Association of Otolaryngologists of India, 1–5. Advance online publication. https://doi.org/10.1007/s12070-021-02689-4

  4. 4.

    Mabrouki FZ, Sekhsoukh R, Aziouaz F, Mebrouk Y (2021) Acute blindness as a complication of severe acute respiratory syndrome Coronavirus-2. Cureus 13(8):e16857 https://doi.org/10.7759/cureus.16857

    PubMed PubMed Central Google Scholar

  5. 5.

    Cyr DG, Vicidomini CM, Siu NY, Elmann SE (2020) Severe bilateral vision loss in 2 patients with coronavirus disease 2019. J Neuroophthalmol 40(3):403–405 https://doi.org/10.1097/WNO.0000000000001039

    Article Google Scholar

  6. 6.

    Atum, M., & Demiryürek, B. E. (2021). Sudden bilateral vision loss in a COVID-19 patient: a case report. Indian J Ophthalmol, 69(8), 2227–2228. https://doi.org/10.4103/ijo. IJO_3706_20

  7. 7.

    Honavar S. G. (2021). Code Mucor: guidelines for the diagnosis, staging and Management of Rhino-Orbito-Cerebral Mucormycosis in the setting of COVID-19. Indian J Ophthalmol, 69(6), 1361–1365. https://doi.org/10.4103/ijo. IJO_1165_21

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Affiliations

  1. A Department Hedi Raies Institute of Ophthalmology, Tunis El-Manar University, Tunis, Tunisie

    Ines Malek, Jihene Sayadi & Leila Nacef

  2. Department of Oto-rhino-laryngology, Charles Nicoles Hospital, Tunis El-Manar University, Tunis, Tunisia

    Rim Lahiani & Memia Ben Salah

  3. Department of Radiology, Charles Nicoles Hospital, Tunis El-Manar University, Tunis, Tunisia

    Miriam Boumediene & Myriam Jrad

  4. Department of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of Monastir, Monastir, Tunisia

    Moncef Khairallah

Authors
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  5. Memia Ben SalahView author publications

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Contributions

IM writing/editing, JS writing/editing, RL editing, MB writing, MBS editing, MJ editing, MK writing/editing, LN editing. The author(s) read and approved the final manuscript.

Corresponding author

Correspondence to Jihene Sayadi.

Ethics approval and consent to participate

This brief report has been performed in accordance with the ethical standards as laid down in 1964 by the declaration of Helsinki and its later amendments. Anonymous case reports are approved by the ethics committee of our institution as long as written consent is obtained from the patients.

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Consent was signed by the patient.

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No conflict of interest.

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Malek, I., Sayadi, J., Lahiani, R. et al. Acute bilateral blindness in a young Covid-19 patient with rhino-orbito-cerebral mucormycosis. J Ophthal Inflamm Infect 11, 40 (2021). https://doi.org/10.1186/s12348-021-00272-0

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中文翻译：
一名年轻的 Covid-19 鼻-眶-脑毛霉菌病患者的急性双侧失明

一名 20 岁糖尿病男性患者之前未接种过 SARS-CoV-2 疫苗，因双侧快速视力丧失伴左侧眶周疼痛、眼球突出、眼睑水肿和肿胀被转诊至我们的急诊科。

一周前，他因 COVID-19 呼吸窘迫综合征在 Covid-19 初级中心入院，并接受了皮质类固醇治疗。

眼科检查，双眼瞳孔固定散大，无光感。左眼表现为眼眶蜂窝织炎的特征（图 1），伴有完全的眼肌麻痹（图 2）。右眼有轻度眼球突出和外展受限（图 2），眼底检查显示视网膜变白，伴有樱桃红色斑点和与视网膜中央动脉阻塞（CRAO）一致的节段性血流（图 3）。扫频源 OCT 显示与缺血性水肿相对应的右眼视网膜内层的高反射（图 3）。

图。1
图1

一名 20 岁的 COVID-19 患者出现鼻眶脑毛霉菌病。( A ) 左侧可见完全性上睑下垂和炎症征象。右侧有轻度眼球突出。( B ) 注意结膜注射、结膜水肿和角膜水肿。( C ) 冠状和 ( D , E) 轴向对比增强脑 CT 扫描显示增厚伴左侧半面部脂肪平面浸润（白星）；左中隔前集合（箭头）与眼球突出和前后轴延长（大箭头）。该集合到达左上颌窦（弯曲箭头）旁边的纤维素脂肪组织，后者是部分液体填充的部位。左侧海绵窦显示血栓形成的迹象（黑色箭头）。（F）轴向 T2 加权图像 MRI 显示额叶区域中的 T2 高信号（大箭头），确认颅内扩展。（G）MR 血管造影显示左颈动脉血栓形成（箭头）。（H）3D T1加权序列对比显示左侧海绵窦血栓形成（弯曲箭头）

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图2
图2

临床照片显示左眼完全麻痹和右眼外展受限

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图 3
图3

( A ) 右眼的眼底摄影显示视网膜变白，有樱桃红点和与视网膜中央动脉闭塞一致的节段血流。( B ) 扫频源 OCT 扫描显示视网膜内层的超反射

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脑部 CT 与脑部 MRI 相辅相成，揭示了疾病的颅内扩展以及左侧海绵窦和左侧颈内动脉的血栓形成（图 1）。

该患者接受了紧急内窥镜鼻窦检查，并从鼻旁窦中取出了黑色坏死组织。组织病理学检查证实了毛霉菌病的诊断。

患者开始接受静脉内脂质体两性霉素 B 和克拉维酸-阿莫西林治疗。地塞米松已停用。与稀释的两性霉素 B 冲洗液一起尝试清创涉及的鼻窦和相邻结构。讨论了眼眶切除术，但由于预后不良和缺乏患者同意而没有保留。

在接下来的几天里，由于双侧广泛的眼睑和面部坏死，左眼球化脓性融化和中枢神经系统受累，患者的眼部和全身状况恶化（图 4）。他在第 30 天去世了。

图 4
图4

初次就诊后 12 天的临床照片显示双侧广泛的眼睑坏死伴左眼球化脓性熔化

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随着 COVID-19 大流行的开始，临床医生发现毛霉菌病病例突然激增。尽管 COVID-19 感染患者可能会累及所有眼部结构，但眼眶毛霉菌病似乎是迄今为止最具侵袭性和致命性的并发症 [1,2,3]。

据我们所知，很少有 Covid-19 患者出现严重双侧失明的病例报告 [2,3,4,5,6]。它们与毛霉菌病的眼眶位置有关 [2, 3] 或与神经系统并发症有关，包括双侧视神经炎 [4] 和枕部缺血性中风 [5, 6]。

我们患者的表现表明左侧海绵窦和颈内动脉血栓形成以及右侧 CRAO。真菌感染从眼动脉和其他眼眶动脉进展到左侧海绵窦、同侧颈动脉和右侧视网膜中央动脉。这导致双侧视力丧失和左侧眼肌麻痹的急性发作。

ROCM的成功治疗很大程度上取决于感染的早期诊断、潜在诱发因素的控制和积极的手术[7]。

因此，眼科医生应该高度怀疑患有 COVID-19 疾病并接受皮质类固醇治疗的糖尿病患者发生毛霉菌病。针对 SARS-COv2 的疫苗接种似乎是预防 COVID-19 和相关致盲和致命疾病的最佳治疗方法。

数据请参考通讯作者。

ROCM：

鼻-眶-脑毛霉菌病

克劳：

视网膜中央动脉闭塞

  1. 1.

    Sen, M., Honavar, SG, Sharma, N., & Sachdev, MS (2021)。COVID-19 和眼睛：对 COVID-19 眼科表现的回顾。印度眼科杂志，69（3），488-509。https://doi.org/10.4103/ijo。IJO_297_21

  2. 2.

    Veisi, A., Bagheri, A., Eshaghi, M., Rikhtehgar, MH, Rezaei Kanavi, M., & Farjad, R. (2021)。COVID-19 患者类固醇治疗期间的鼻眼眶毛霉菌病：病例报告。欧洲眼科杂志，11206721211009450。提前在线出版。https://doi.org/10.1177/11206721211009450, 112067212110094

  3. 3.

    Eswaran, S.、Balan, SK 和 Saravanam, PK (2021)。由 Covid 19 感染引发的年轻患者的急性暴发性毛霉菌病。印度耳鼻喉科和头颈外科杂志：印度耳鼻喉科医师协会的官方出版物，1-5。提前在线发布。https://doi.org/10.1007/s12070-021-02689-4

  4. 4.

    Mabrouki FZ、Sekhsoukh R、Aziouaz F、Mebrouk Y（2021）作为严重急性呼吸系统综合症冠状病毒 2 的并发症的急性失明。Cureus 13(8):e16857 https://doi.org/10.7759/cureus.16857

    PubMed PubMed Central Google Scholar

  5. 5.

    Cyr DG, Vicidomini CM, Siu NY, Elmann SE (2020) 2 名冠状病毒病患者严重双侧视力丧失 2019. J Neuroophthalmol 40(3):403–405 https://doi.org/10.1097/WNO.000000000000103

    文章 谷歌学术

  6. 6.

    Atum, M. 和 Demiryürek, BE (2021)。COVID-19 患者双侧视力突然丧失：病例报告。印度眼科杂志，69（8），2227-2228。https://doi.org/10.4103/ijo。IJO_3706_20

  7. 7.

    霍纳瓦尔 (2021)。Code Mucor：在 COVID-19 环境中诊断、分期和管理 Rhino-Orbito-Cerebral Mucormycosis 的指南。印度眼科杂志，69（6），1361-1365。https://doi.org/10.4103/ijo。IJO_1165_21

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  1. A Department Hedi Raies 眼科研究所，突尼斯 El-Manar 大学，突尼斯，突尼斯

    伊内斯·马利克、吉赫内·萨亚迪和莱拉·纳塞夫

  2. 突尼斯 El-Manar 大学 Charles Nicoles 医院耳鼻喉科，突尼斯突尼斯

    Rim Lahiani & Memia Ben Salah

  3. 突尼斯 El-Manar 大学 Charles Nicoles 医院放射科，突尼斯，突尼斯

    米里亚姆·布迈丁和米里亚姆·杰拉德

  4. Fattouma Bourguiba 大学医院眼科，莫纳斯提尔大学医学院，莫纳斯提尔，突尼斯

    蒙塞夫·海拉拉

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Malek, I.、Sayadi, J.、Lahiani, R.等。一名患有鼻眼眶脑毛霉菌病的年轻 Covid-19 患者的急性双侧失明。J Ophthal Inflamm Infect 11, 40 (2021)。https://doi.org/10.1186/s12348-021-00272-0

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