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导致颞下颌关节强直的鼻-眶-脑型毛霉菌病:两例病例系列报道

Rhino-Orbital Cerebral Mucormycosis Causing Temporomandibular Joint Ankylosis: A Case Series of Two Patients.

作者信息

Mokhtar Ejaz A, Fatima Qalbi, Akbar Shahrukh, Equbal Sharique, Salahudeen Ameera

机构信息

Oral and Maxillofacial Surgery, All India Institute of Medical Sciences, Patna, IND.

Periodontics, Buddha Institute of Dental Sciences and Hospital, Patna, IND.

出版信息

Cureus. 2023 Feb 19;15(2):e35194. doi: 10.7759/cureus.35194. eCollection 2023 Feb.

DOI:10.7759/cureus.35194
PMID:36960262
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10030649/
Abstract

Temporomandibular joint ankylosis is caused by trauma, infection, autoimmune, inflammatory joint diseases, and several other minor causes. Mucormycosis causing temporomandibular joint ankylosis has not been reported. We report two cases of temporomandibular joint ankylosis caused by mucormycosis from 2018 to 2022. In both cases, the infection started in the maxilla, then progressed to orbit. After that, involvement of the mastoid process, styloid process, and base of the skull was observed in the first case, while in the second case, there was the involvement of the base of the skull and mandibular ramus. As the temporomandibular joint (TMJ) components are contiguous to the base of the skull, it got affected causing temporomandibular joint ankylosis. Mucormycosis was diagnosed by KOH mount. The smear examination showed aseptate hyphae at 90. Histopathology examination further confirmed mucormycosis. Glycemic control was done by infusing Insulin (both Lantus and regular). The case was managed with aggressive debridement and interpositional arthroplasty with a buccal fat pad. Liposomal amphotericin infusion was also started pre-operatively and continued in the post-operative phase. After 4 years of follow-up, the patient is well and had adequate mouth opening. Mucormycosis infection affecting the TMJ has been reported in the literature. However, this is the first report of a mucormycosis infection resulting in TMJ ankylosis in the literature. The infection should be aggressively managed. Reversals of an immunocompromised state, aggressive surgical management, and antifungal medication are the key factors for the success of the deadly fungal infection.

摘要

颞下颌关节强直由创伤、感染、自身免疫性、炎性关节疾病以及其他一些次要原因引起。由毛霉菌病导致颞下颌关节强直的情况尚未见报道。我们报告了2018年至2022年期间由毛霉菌病引起的两例颞下颌关节强直病例。在这两例中,感染均始于上颌,随后蔓延至眼眶。之后,在第一例中观察到乳突、茎突和颅底受累,而在第二例中,颅底和下颌支受累。由于颞下颌关节(TMJ)各组成部分与颅底相邻,因而受到影响导致颞下颌关节强直。通过氢氧化钾涂片诊断为毛霉菌病。涂片检查显示90°的无隔菌丝。组织病理学检查进一步证实为毛霉菌病。通过输注胰岛素(来得时和常规胰岛素)进行血糖控制。该病例采用积极清创术和颊脂垫间置关节成形术进行治疗。术前也开始输注脂质体两性霉素,并在术后阶段继续使用。经过4年的随访,患者情况良好,开口度正常。文献中已报道毛霉菌病感染影响颞下颌关节。然而,这是文献中首例毛霉菌病感染导致颞下颌关节强直的报告。应对该感染进行积极治疗。逆转免疫功能低下状态、积极的手术治疗和抗真菌药物治疗是成功治疗这种致命真菌感染的关键因素。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/1800ec15fb20/cureus-0015-00000035194-i07.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/fbbacd588f1a/cureus-0015-00000035194-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/73847ce9fe51/cureus-0015-00000035194-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/005ec754dc0c/cureus-0015-00000035194-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/61214e1ff2ae/cureus-0015-00000035194-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/9aa499a079f6/cureus-0015-00000035194-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/67973e4febbd/cureus-0015-00000035194-i06.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/1800ec15fb20/cureus-0015-00000035194-i07.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/fbbacd588f1a/cureus-0015-00000035194-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/73847ce9fe51/cureus-0015-00000035194-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/005ec754dc0c/cureus-0015-00000035194-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/61214e1ff2ae/cureus-0015-00000035194-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/9aa499a079f6/cureus-0015-00000035194-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/67973e4febbd/cureus-0015-00000035194-i06.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d4fa/10030649/1800ec15fb20/cureus-0015-00000035194-i07.jpg

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本文引用的文献

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Mandibular Mucormycosis: A Report of Four Cases and a Discussion on Their Management.下颌毛霉菌病:4例报告及其治疗探讨
Cureus. 2022 Oct 14;14(10):e30301. doi: 10.7759/cureus.30301. eCollection 2022 Oct.
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A Rare Case of Mandibular Mucormycosis in a Post-COVID-19 Patient.一名新冠后患者罕见的下颌骨毛霉菌病病例
Cureus. 2022 Aug 20;14(8):e28216. doi: 10.7759/cureus.28216. eCollection 2022 Aug.
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Granulomatous Mucormycosis of the Temporal Bone extending Into Temporomandibular Joint and Infratemporal Fossa: A Case Report.
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