Gaelic Tongue 4 Letters
Gaelic Tongue 4 Letters – Sub Specialty Fellow, Division of Rheumatology, Department of Pediatrics, British Columbia (BC) Children’s Hospital, University of British Columbia;
Assistant Clinical Professor, Department of Pediatrics and Associate Member, Department of Dermatology and Skin Sciences, University of British Columbia;
Gaelic Tongue 4 Letters
Clinical Associate Professor of Pediatrics, Division of Dermatology, BC Children’s Hospital, University of British Columbia, Vancouver, British Columbia, Canada.
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Parry-Romberg syndrome (PRS) is a rare disease characterized by progressive hematrophy of the skin and soft tissues of the face. It can also result in the atrophy of deeper structures, such as muscles, cartilage, and bones
. We describe a surprising involvement of the mucous membrane of the tongue in 2 girls with PRS. The cases came from the British Columbia Children’s Hospital.
A 4-year-old female had hyperpigmentation on her right chin for 6 months. Examination revealed hyperpigmented plaques over the right ear, behind the right ear extending to the jaw, and over the mental prominence. The lobe and helix of the right ear were atrophic. Oral examination revealed a deviation of the tongue to the right and a hematrophy with loss of papillae (Figure 1, left panel). Anti-nuclear, anti-dsDNA and antibody tests
They were negative. Treatment with oral prednisone 5 mg twice daily and 15 mg oral methotrexate (MTX) weekly resulted in no progression of lesions over 2 years.
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Left panel: Right-sided tongue hemiatrophy in a 4-year-old girl. Right panel: Hemiatrophy and loss of right side tongue papillae in a 15-year-old female.
A 15-year-old woman presented with hypopigmentation and induration of the right paranasal skin. Physical examination revealed hardened plaque along the right paranasal skin with atrophic plaques extending from the right cheek to the chin and neck. Oral examination revealed hemiatrophy and loss of dermal papillae on the right side of the tongue (Figure 1, right panel). Treatment with oral prednisone 15 mg twice daily and oral MTX 20 mg weekly was initiated with partial softening and resolution of facial and neck lesions. Oral lesions showed minimal change during the 4-year follow-up. A 79-year-old woman was referred to her general practitioner for an urgent surgical revision of a necrotic tongue. She had been fatigued for the past 3 weeks and complained of bilateral throat and occipital neck pain which progressed to include dental pain, jaw claudication and difficulty opening her mouth. She denied the visual symptoms, but this was complicated by a background of cataracts and macular degeneration. However, she believed that her vision had deteriorated during this illness. She had no allergies and oral trauma; the tongue had become necrotic 4 days before the revision.
On examination she was dysphagic; the tongue was clearly demarcated between healthy and necrotic tissue, painful with speech and touch, insensitive to taste and produced a putrid odor. A literature search revealed that this is an extremely rare manifestation of GCA
. She was given intravenous ticarcillin / clavulanate every 6 hours and received mouthwashes every 8 hours. Blood tests revealed an erythrocyte sedimentation rate of 75 mm / h. Discussion with the rheumatology specialist led to immediate empiric steroid treatment. Steroid treatment resulted in an almost immediate arrest of necrosis; a subsequent temporal artery biopsy confirmed GCA.
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After one week of treatment, there was a significant improvement in sensation and speech, with the necrotic segment of the tongue completely self-amputating (Figure 2). The patient has since been followed up by the rheumatology service and continues to improve. A paucity of similar cases in the literature has followed a comparable course
. Physicians are advised to pay attention to this rare manifestation of GCA, allowing for prompt steroid therapy and the involvement of a rheumatology specialist.
Recession of pain and symptoms and self-amputation of the necrotic segment of the tongue after 1 week of steroid treatment.Julia A. Hunter, MB BCh BAO; Deborah M. Levy, MD, MS, FRCPC; Roberta A. Berard, MD, MSc, FRCPC, Division of Pediatric Rheumatology, Western University, London, Ontario, Canada.
. We are not aware of the previously described lingual involvement in individuals with primary PR, particularly children.
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A previously healthy 6-year-old prepubertal female was referred for rheumatology evaluation. After exposure to cold, she described a typical three-phase RP of the fingers. After initial episodes involving the fingers, she subsequently experienced painless blanching of the left anterior third of the tongue associated with tingling. These episodes were precipitated by exposure to cold with the mouth open, but not by the ingestion of cold liquids or foods. The exact duration of the episode is unknown, although rapid resolution occurred after closing the mouth. There was no history of lingual ulcer or other associated symptoms. Lingual and digital episodes did not occur simultaneously (Figures 1A, B).
A 6-year-old female with a history of blanching and tingling fingers developed predominantly left anterior tongue discoloration consistent with lingual RP. (A) upper surface of the tongue. (B) Typical finger bleaching as seen in RP. RP: Raynaud’s phenomenon.
History and examination, including nail capillaroscopy, were normal. Laboratory analysis revealed positive antinuclear antibodies (ANA; titer of 1: 2560 in a mitotic pattern). The extractable nuclear antigen (ENA) panel was negative. The patient was evaluated 9 months later, with repeated ANAs revealing a titer of 1: 160 (mitotic). ENA remained negative. There was no symptom evolution suggesting secondary RP.
Retrospective data have shown that ANA positivity in children with RP is associated with evolution to involve other features of a systemic autoimmune disease
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. Although she had no secondary RP at the time of evaluation, given this patient’s elevated ANA and unusual lingual presentation, she will be closely monitored for the development of associated rheumatic disease. The history of Clan Gregor is as turbulent as the history of Scotland itself. Claiming ancestry from the first Scottish king, Alpin, through his second son, Griogor, the motto of Clan McGregor of ‘S rioghal mo dhream translates from the Gaelic language as “Royal is my race” and reflects this heritage.
From the beginning the McGregors were a scattered people and took many names, but collectively they were known as “the Gregorachs”. They have maintained extremely close family ties and loyalties despite their fractured state and this more than anything else would have been of use to them in the latter part of the second millennium.
Perhaps the greatest misfortune for this clan has been to occupy their traditional homelands, lands coveted by the nearby and powerful Clan Campbell. This clan house was the eastern border of Argyll and the western border of Perthshire, including Glenorchy, Glenstrae, Glenlyon and Glengyle. The clan’s first possession, Glenorchy which, previously owned by the Campbells, was bestowed on the McGregors for services rendered to King Alexander II (1214-1249) in his conquest of Argyll. This undoubtedly earned them the enmity of Clan Campbell in a country where feuds between families and clans lasted for generations and were rather bloody affairs. For a long time the McGregors retained possession of their lands by right of the sword, however the enmity of the surrounding clans led to attempts to overthrow the clan and the inevitable retaliation by the McGregors, which thus earned a reputation as a turbulent people.
Certainly the first recognized head of the Gregor Clan was Griogair (Gregor) of the Golden Bridles, born around 1300 and died around 1360.
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By 1440 the Campbells had gained control of the Glenorchy lands by expelling McGregor and other tenants, replacing them with Campbell. When Iain Dubh of the house of Glenstrae died in 1519, he left no heirs. Moving over the older houses of Brackley and Roro, the McGregor’s superiors (landowners), the Glenorchy Campbells, enforced the succession of Eoin MacEoghan, head of the line known as Clan Dughaill Ciar and resident of Glengyle, as McGregor’s 7th chief. . This was because Eoin had once raped and married the daughter of Sir Colin Campbell of Glenorchy, who now hoped to control Gregorach through his son-in-law. These McGregors turned out to be the most undisciplined of all.
The McGregors were fair game for any landowner who possessed the strength and fortitude to stand up to them. They were largely dispersed due to the Campbells ‘greed, but later became pawns on the Campbells’ expansion agenda.
Sir Humphrey Colquhoun, the Laird of Luss was also a greedy and greedy laird, who became a nuisance to the Campbells. The most powerful of the laird Campbell was the Duke of Argyll. He tacitly directed the McGregors to Luss to reduce his northward expansion into Campbell’s territory. As the hereditary high Sheriff of Scotland, Argyll had all the credentials in this fight. If the blame were to go around, Argyll would fall on the author, be it McGregor or Luss. With his influence and his prestige at court, Argyll kept an eye on the proceedings.
In truth, the McGregors were a fairly illegal clan, which could organize raids on the plains and territories of neighboring clans. This they did, being extremely good restaurateurs or cattle ranchers (cattle thieves), and maintained a very successful blackmail enterprise by “protecting” the cattle and other assets of their “customers”. They could, when needed, retreat safely behind the highland fortress of their mountains, valleys and friendly families.
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The Laird of Luss saw an opportunity to gain lands from Clan Gregor in battle and began actively harassing all McGregors that came within their reach. Two of these itinerant Gregorach men,