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Sialo-cutaneous fistula with ectopic submandibular gland sialolith, revealing a hidden ipsilateral enlarged and elongated styloid process: a consideration based on CT findings

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Abstract

Sialolithiasis is a common salivary pathology, and an uncommon complication of sialadenitis and sialolithiasis is the formation of fistulous tracts to other compartments. Submandibular gland sialo-oral fistulae are not particularly remarkable, given the location of the gland and Wharton’s duct, but submandibular sialolith-associated fistulae to other cervico-facial compartments (transcervical sialo-cutaneous and sialo-pharyngeal fistulae) are much less common. We report herein an unusual case of a 49-year-old obese man with sialo-cutaneous fistula containing a large, ectopic sialolith in subcutaneous tissue that was expected to undergo spontaneous elimination, but revealed hidden Eagle syndrome featuring an ipsilateral enlarged, elongated styloid process. Furthermore, we offer a thorough review of the literature regarding sialo-fistulae and highlight the relationship between an abnormal styloid process and submandibular sialadenitis with sialolithiasis and new tract formation based on computed tomography.

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Acknowledgements

This work was supported by the Private University Research Branding Project from MEXT of Japan.

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Authors and Affiliations

  1. Department of Oral and Maxillofacial Radiology, Tokyo Dental College, Tokyo, Japan

    Mamoru Wakoh & Tazuko K. Goto

  2. Division of Dental Radiology, Tokyo Dental College Chiba Dental Care Center, 1-2-2 Masago, Mihama-ku, Chiba-City, Chiba, 261-8502, Japan

    Mamoru Wakoh

  3. Department of Pathology, Tokyo Dental College, Tokyo, Japan

    Kenichi Matsuzaka

  4. Department of Oral and Maxillofacial Surgery, Tokyo Dental College, Tokyo, Japan

    Takahiko Shibahara

  5. Department of Oral and Maxillofacial Radiology, The Nippon Dental University, Tokyo, Japan

    Takashi Kamio

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  1. Mamoru Wakoh
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  2. Tazuko K. Goto
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  4. Takahiko Shibahara
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Correspondence to Mamoru Wakoh.

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Authors M. Wakoh, TK. Goto, T. Shibahara, K Matsuzaka and T Kamio declare that they have no conflict of interest.

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All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation and with the Helsinki Declaration of 1964 and later versions. Informed consent was obtained from a patient for being included in the study. Additional informed consent was obtained from a patient for which identifying information is included in this article.

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Wakoh, M., Goto, T.K., Matsuzaka, K. et al. Sialo-cutaneous fistula with ectopic submandibular gland sialolith, revealing a hidden ipsilateral enlarged and elongated styloid process: a consideration based on CT findings. Oral Radiol 37, 336–344 (2021). https://doi.org/10.1007/s11282-020-00481-8

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  • DOI: https://doi.org/10.1007/s11282-020-00481-8

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