Intra-parenchymal sialolithiasis and subsequent fibrosis of the submandibular salivary glands is a rare disorder. The resulting swelling, pain, and infection derives affected patients to seek treatment. We present the case of an 85-years-old Saudi male patient who suffered from repeated swelling and infection in the left submandibular region which was misdiagnosed and treated for over 60 years as dental infection, infected skin sebaceous gland or lipoma. The presented case represents the largest intra-glandular submandibular stone with the longest duration ever reported in the medical literature.
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