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Aplasia is not the cause of missing submandibular gland
International Journal of Surgery Case Reports 4 (2013) 540
Contents lists available at SciVerse ScienceDirect
International Journal of Surgery Case Reports journal homepage: www.elsevier.com/locate/ijscr
Letter to the Editor Aplasia is not the cause of missing submandibular gland A recent article described the management of a recurrent oral ranula over a period of 6 months when a CT scan was performed and showed the ranula with absence of the ipsilateral submandibular gland.1 This was interpreted to indicate unilateral aplasia of the submandibular gland. However, this is unlikely, and there is another, likely explanation for the absence of the submandibular gland that relates to the treatment of the ranula. The oral ranula was treated by excision and recurred several times. A ranula is an extravasation mucocele, which was confirmed histologically in the present case,1 and is resorbed when the source of mucus, namely the sublingual gland, is removed.2–4 Excision of oral ranula alone has been found in a literature review to have been unsuccessful in 50% of cases excluding postnatal ranula, and moreover, treatment by excision of the ranula subjects the patient to unnecessary surgery.2,3 The ranula in the present case was eventually treated successfully by excision of the sublingual gland.1 In a previously investigated case of recurrent oral ranula, the disappearance of the ipsilateral submandibular gland was seen to occur over a period of 5 months following excision of the ranula and associated sublingual gland with damage to Wharton’s duct.5 The leaking Wharton’s duct became sealed by postoperative fibrosis, which caused complete obstruction of the submandibular gland, which has much less resistance to complete obstruction than the sublingual gland and atrophies.2,3,5 This is clearly what happened in the present case during the 6 months between initial surgery and the CT scan. The ranula in the present case was found at final surgery to communicate with a blindly ending duct,1 which was the Wharton’s duct that had been completely obstructed by postoperative fibrosis. A similar misinterpretation of a vanished submandibular gland occurred in two cases in which a sialolith had caused complete obstruction and atrophy of the submandibular gland.6 Unfortunately there is no shortage of misinformation in the recent article,1 such as: the statements about the rate of recurrence of oral ranulas with reference to Zhao et al. are incorrect because Zhao et al. considered oral, plunging and mixed ranulas together and did not differentiate oral ranulas7,8 ; the statement that the rate of recurrence of oral ranula treated by complete excision of the ranula is up to 25% is not referenced, is incorrect, and appears to be no more than imaginary; the statement that oral ranula is a retention cyst arising as a result of ductal obstruction of the sublingual gland is incorrect because oral ranula is an extravasation cyst arising from damage to a duct of the sublingual gland or rarely from obstruction of the sublingual gland that leads to rupture of acini.2,3,9,10
In conclusion, when faced with a missing submandibular gland, seek a cause for obstruction and complete atrophy, which cause the submandibular gland to vanish. Conflicts of interest None. Funding None. References 1. Albsoul NM, Obeidat FO, Altaher RN, Jubouri SA, Hadidy AM. Recurrent right sublingual ranula, concomitant with ipsilateral submandibular salivary gland aplasia. International Journal of Surgery Case Reports 2013;4:229–31. 2. Harrison JD. Modern management and pathophysiology of ranula: literature review. Head and Neck 2010;32:1310–20. 3. Harrison JD. Aetiology and treatment of ranula. Journal of ENT Masterclass 2010;3:22–5. 4. Samant S, Morton RP, Ahmad Z. Surgery for plunging ranula: the lesson not yet learned? European Archives of Oto-Rhino-Laryngology and Head & Neck 2011;268:1513–8. 5. Harrison JD, Sowray JH, Smith NJD. Recurrent ranula. A case report. British Dental Journal 1976;140:180–2. 6. Harrison JD. Letter to the editor: Sialolithiasis in a stump of Wharton’s duct of an aplastic unilateral submandibular gland. International Journal of Oral and Maxillofacial Surgery 2009;38:1344. 7. Zhao Y-F, Jia Y, Chen X-M, Zhang W-F. Clinical review of 580 ranulas. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology and Endodontology 2004;98:281–7. 8. Zhao Y-F, Jia J, Jia Y. Complications associated with surgical management of ranulas. Journal of Oral and Maxillofacial Surgery 2005;63:51–4. 9. McGurk M, Eyeson J, Thomas B, Harrison JD. Conservative treatment of oral ranula by excision with minimal excision of the sublingual gland: histological support for a traumatic etiology. Journal of Oral and Maxillofacial Surgery 2008;66:2050–7. 10. Harrison JD. Salivary mucoceles. Oral Surgery, Oral Medicine, Oral Pathology 1975;39:268–78.
John D. Harrison ∗ King’s College London Dental Institute at Guy’s, King’s College and St Thomas’ Hospitals, London, United Kingdom ∗ Correspondence
address: Unit of Oral Pathology, Floor 28, Tower Wing, Guy’s Hospital, London, SE1 9RT, United Kingdom. E-mail address: [email protected]
Available online 21 March 2013
Open Access This article is published Open Access at sciencedirect.com. It is distributed under the IJSCR Supplemental terms and conditions, which permits unrestricted non commercial use, distribution, and reproduction in any medium, provided the original authors and source are credited. 2210-2612/$ – see front matter © 2013 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijscr.2013.03.006
Contents lists available at SciVerse ScienceDirect
International Journal of Surgery Case Reports journal homepage: www.elsevier.com/locate/ijscr
Letter to the Editor Aplasia is not the cause of missing submandibular gland A recent article described the management of a recurrent oral ranula over a period of 6 months when a CT scan was performed and showed the ranula with absence of the ipsilateral submandibular gland.1 This was interpreted to indicate unilateral aplasia of the submandibular gland. However, this is unlikely, and there is another, likely explanation for the absence of the submandibular gland that relates to the treatment of the ranula. The oral ranula was treated by excision and recurred several times. A ranula is an extravasation mucocele, which was confirmed histologically in the present case,1 and is resorbed when the source of mucus, namely the sublingual gland, is removed.2–4 Excision of oral ranula alone has been found in a literature review to have been unsuccessful in 50% of cases excluding postnatal ranula, and moreover, treatment by excision of the ranula subjects the patient to unnecessary surgery.2,3 The ranula in the present case was eventually treated successfully by excision of the sublingual gland.1 In a previously investigated case of recurrent oral ranula, the disappearance of the ipsilateral submandibular gland was seen to occur over a period of 5 months following excision of the ranula and associated sublingual gland with damage to Wharton’s duct.5 The leaking Wharton’s duct became sealed by postoperative fibrosis, which caused complete obstruction of the submandibular gland, which has much less resistance to complete obstruction than the sublingual gland and atrophies.2,3,5 This is clearly what happened in the present case during the 6 months between initial surgery and the CT scan. The ranula in the present case was found at final surgery to communicate with a blindly ending duct,1 which was the Wharton’s duct that had been completely obstructed by postoperative fibrosis. A similar misinterpretation of a vanished submandibular gland occurred in two cases in which a sialolith had caused complete obstruction and atrophy of the submandibular gland.6 Unfortunately there is no shortage of misinformation in the recent article,1 such as: the statements about the rate of recurrence of oral ranulas with reference to Zhao et al. are incorrect because Zhao et al. considered oral, plunging and mixed ranulas together and did not differentiate oral ranulas7,8 ; the statement that the rate of recurrence of oral ranula treated by complete excision of the ranula is up to 25% is not referenced, is incorrect, and appears to be no more than imaginary; the statement that oral ranula is a retention cyst arising as a result of ductal obstruction of the sublingual gland is incorrect because oral ranula is an extravasation cyst arising from damage to a duct of the sublingual gland or rarely from obstruction of the sublingual gland that leads to rupture of acini.2,3,9,10
In conclusion, when faced with a missing submandibular gland, seek a cause for obstruction and complete atrophy, which cause the submandibular gland to vanish. Conflicts of interest None. Funding None. References 1. Albsoul NM, Obeidat FO, Altaher RN, Jubouri SA, Hadidy AM. Recurrent right sublingual ranula, concomitant with ipsilateral submandibular salivary gland aplasia. International Journal of Surgery Case Reports 2013;4:229–31. 2. Harrison JD. Modern management and pathophysiology of ranula: literature review. Head and Neck 2010;32:1310–20. 3. Harrison JD. Aetiology and treatment of ranula. Journal of ENT Masterclass 2010;3:22–5. 4. Samant S, Morton RP, Ahmad Z. Surgery for plunging ranula: the lesson not yet learned? European Archives of Oto-Rhino-Laryngology and Head & Neck 2011;268:1513–8. 5. Harrison JD, Sowray JH, Smith NJD. Recurrent ranula. A case report. British Dental Journal 1976;140:180–2. 6. Harrison JD. Letter to the editor: Sialolithiasis in a stump of Wharton’s duct of an aplastic unilateral submandibular gland. International Journal of Oral and Maxillofacial Surgery 2009;38:1344. 7. Zhao Y-F, Jia Y, Chen X-M, Zhang W-F. Clinical review of 580 ranulas. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology and Endodontology 2004;98:281–7. 8. Zhao Y-F, Jia J, Jia Y. Complications associated with surgical management of ranulas. Journal of Oral and Maxillofacial Surgery 2005;63:51–4. 9. McGurk M, Eyeson J, Thomas B, Harrison JD. Conservative treatment of oral ranula by excision with minimal excision of the sublingual gland: histological support for a traumatic etiology. Journal of Oral and Maxillofacial Surgery 2008;66:2050–7. 10. Harrison JD. Salivary mucoceles. Oral Surgery, Oral Medicine, Oral Pathology 1975;39:268–78.
John D. Harrison ∗ King’s College London Dental Institute at Guy’s, King’s College and St Thomas’ Hospitals, London, United Kingdom ∗ Correspondence
address: Unit of Oral Pathology, Floor 28, Tower Wing, Guy’s Hospital, London, SE1 9RT, United Kingdom. E-mail address: [email protected]
Available online 21 March 2013
Open Access This article is published Open Access at sciencedirect.com. It is distributed under the IJSCR Supplemental terms and conditions, which permits unrestricted non commercial use, distribution, and reproduction in any medium, provided the original authors and source are credited. 2210-2612/$ – see front matter © 2013 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijscr.2013.03.006