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Giant Cystic Dilatation That Includes Multiple Sialolithiasis of Submandibular Gland
Otolaryngology–Head and Neck Surgery (2006) 134, 533-534
CASE REPORT
Giant Cystic Dilatation That Includes Multiple Sialolithiasis of Submandibular Gland M. Engin Uluç, MD, Berna Dirim Vidinli, MD, Nezahat Erdogˇan, MD, and Fazıl Gelal, MD, Izmir, Turkey
S
ialolithiasis is the formation of calcific concretions in the salivary duct or glands.1 Most calculi occur in the submandibular gland. The cause is unclear; inflammation is the widely accepted causative condition. Sialolithiasis may also be the initiating cause of sialoadenitis. In some of the cases objective findings are subtle but may be dramatic in others. Pain and swelling are the most frequent symptoms. Diagnosis is frequently made after an acute obtructive and/or inflammatory episode.2 Ultrasonography, radiograph examinations, and computed tomography can be used for diagnosis and MR sialography with T2W fast SE is a new and sophisticated technique.3,4 Differential diagnosis must be done with obstructive sialadenitis, epidemic parotitis, and tumors of the salivar gland.4,5 Treatment is surgical removal of the calculi. Noninvasive new techniques such as extracorporeal shock wave lithotripsy (ESWL) and endoscopic ıntracorporeal lithotripsy (ESIL) can be applied when available. Occasionally spontaneous extrusion of the calculus through the ductal orifice may occur. Extirpation of the entire gland may be the last resort for the untreatable calculi. This article describes an unusual case of chronic submandibular sialolithiasis with multiple calculu and giant cystic dilatation of the gland. A 37-year-old mentally retarded man presented with painless recurrent right submandibular swelling of 5 years duration. He could not express any personal complaints so no From the Department of Radiology, I˙zmir Atatürk Education and Investigation Hospital, Yes¸ilyurt/I˙zmir, Turkey Reprint requests Dr Berna Dirim Vidinli, I˙zmir Atatürk Education ve Investigation Hospital Department of Radiology, 35360 Izmir, Turkey.
therapeutic procedures had ever been applied. Clinical examination revealed a soft palpable painless mass in the right submandibular space. No lymphadenopathy was noted. Ultrasonography was perfomed with a 7.5 MHz linear probe and multiple reflective-mobile structures were observed in the lesion. CT examination was performed in the prone and supine positions. A giant cystic pouch with multiple calculi in the submandibular gland location was seen. The submandibular gland itself could not be observed. Calculi were dependently mobile (Fig 1). The diagnosis in our case is chronic sialolithiasis, submandibular gland atrophy with cystic dilatation of the submandibuar duct/gland. After a short period of supportive treatment the right submandibular gland was excised.
CONCLUSIONS This case is interesting because of the giant cystic dilatation of the glandular component and also because of the unusual number and dimensions of the calculi. This result that can only be achieved if the patient is not aware of his/her own illness. Early diagnosis can bring the urgent and correct solutions in such conditions, and this is not a problem for today’s technologies. However, the mental retardation of the patient and the probable undereducation of the his parents postponed the treatment and caused the exaggerated pathology. E-mail address: [email protected].
0194-5998/$32.00 © 2006 American Academy of Otolaryngology–Head and Neck Surgery Foundation, Inc. All rights reserved. doi:10.1016/j.otohns.2005.03.073
534
Otolaryngology–Head and Neck Surgery, Vol 134, No 3, March 2006
REFERENCES 1. Karengera D, Yousefpour A, Reychler H. Unusual elimination of a salivary calculus. Int J Oral Maxillofac Surg 1998;27:224 –5. 2. Haring JH. Diagnosing salivary stones. J Am Dent Assoc 1990;122: 75– 6. 3. Lomas DJ, Carroll MR, Johnson G, et al. MR sialography: work in progress. Radiology 1996;200:129 –33. 4. Fishbach R, Kugel H, Exast E, et al. MR sialography: initial experience using a T2 weighted fast SE sequence. J Comput Assist Tomogr 1997; 21:826 –30. 5. Rice DH, Salivary gland disorders: neoplastic and nonneoplastic. Med Clin North Am 1999;83:197–218.
Figure 1 A, Axial CT scan in supine position shows multiple calculis in a giant cystic cavity in the submandibular gland. B, Axial CT scan of the same level in prone position shows calculi gathered at the bottom of the cystic cavity.
CASE REPORT
Giant Cystic Dilatation That Includes Multiple Sialolithiasis of Submandibular Gland M. Engin Uluç, MD, Berna Dirim Vidinli, MD, Nezahat Erdogˇan, MD, and Fazıl Gelal, MD, Izmir, Turkey
S
ialolithiasis is the formation of calcific concretions in the salivary duct or glands.1 Most calculi occur in the submandibular gland. The cause is unclear; inflammation is the widely accepted causative condition. Sialolithiasis may also be the initiating cause of sialoadenitis. In some of the cases objective findings are subtle but may be dramatic in others. Pain and swelling are the most frequent symptoms. Diagnosis is frequently made after an acute obtructive and/or inflammatory episode.2 Ultrasonography, radiograph examinations, and computed tomography can be used for diagnosis and MR sialography with T2W fast SE is a new and sophisticated technique.3,4 Differential diagnosis must be done with obstructive sialadenitis, epidemic parotitis, and tumors of the salivar gland.4,5 Treatment is surgical removal of the calculi. Noninvasive new techniques such as extracorporeal shock wave lithotripsy (ESWL) and endoscopic ıntracorporeal lithotripsy (ESIL) can be applied when available. Occasionally spontaneous extrusion of the calculus through the ductal orifice may occur. Extirpation of the entire gland may be the last resort for the untreatable calculi. This article describes an unusual case of chronic submandibular sialolithiasis with multiple calculu and giant cystic dilatation of the gland. A 37-year-old mentally retarded man presented with painless recurrent right submandibular swelling of 5 years duration. He could not express any personal complaints so no From the Department of Radiology, I˙zmir Atatürk Education and Investigation Hospital, Yes¸ilyurt/I˙zmir, Turkey Reprint requests Dr Berna Dirim Vidinli, I˙zmir Atatürk Education ve Investigation Hospital Department of Radiology, 35360 Izmir, Turkey.
therapeutic procedures had ever been applied. Clinical examination revealed a soft palpable painless mass in the right submandibular space. No lymphadenopathy was noted. Ultrasonography was perfomed with a 7.5 MHz linear probe and multiple reflective-mobile structures were observed in the lesion. CT examination was performed in the prone and supine positions. A giant cystic pouch with multiple calculi in the submandibular gland location was seen. The submandibular gland itself could not be observed. Calculi were dependently mobile (Fig 1). The diagnosis in our case is chronic sialolithiasis, submandibular gland atrophy with cystic dilatation of the submandibuar duct/gland. After a short period of supportive treatment the right submandibular gland was excised.
CONCLUSIONS This case is interesting because of the giant cystic dilatation of the glandular component and also because of the unusual number and dimensions of the calculi. This result that can only be achieved if the patient is not aware of his/her own illness. Early diagnosis can bring the urgent and correct solutions in such conditions, and this is not a problem for today’s technologies. However, the mental retardation of the patient and the probable undereducation of the his parents postponed the treatment and caused the exaggerated pathology. E-mail address: [email protected].
0194-5998/$32.00 © 2006 American Academy of Otolaryngology–Head and Neck Surgery Foundation, Inc. All rights reserved. doi:10.1016/j.otohns.2005.03.073
534
Otolaryngology–Head and Neck Surgery, Vol 134, No 3, March 2006
REFERENCES 1. Karengera D, Yousefpour A, Reychler H. Unusual elimination of a salivary calculus. Int J Oral Maxillofac Surg 1998;27:224 –5. 2. Haring JH. Diagnosing salivary stones. J Am Dent Assoc 1990;122: 75– 6. 3. Lomas DJ, Carroll MR, Johnson G, et al. MR sialography: work in progress. Radiology 1996;200:129 –33. 4. Fishbach R, Kugel H, Exast E, et al. MR sialography: initial experience using a T2 weighted fast SE sequence. J Comput Assist Tomogr 1997; 21:826 –30. 5. Rice DH, Salivary gland disorders: neoplastic and nonneoplastic. Med Clin North Am 1999;83:197–218.
Figure 1 A, Axial CT scan in supine position shows multiple calculis in a giant cystic cavity in the submandibular gland. B, Axial CT scan of the same level in prone position shows calculi gathered at the bottom of the cystic cavity.