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Rapid Communication| Volume 133, ISSUE 2, P129-137, February 2022

Ill-defined extensive radiolucent lesion of the left posterior mandible

      A 60-year-old female patient was referred by an outside oral and maxillofacial surgeon for evaluation of a left posterior mandibular “lesion.” The patient estimated that she had first experienced pain associated with the left mandibular posterior teeth approximately 6 months before presentation. She also experienced progressive difficulty with mastication on the left side. She was seen by her general dentist and was referred to an oral surgeon for extraction of the left mandibular posterior teeth due to periodontally compromised dentition. After extraction of the left posterior molars, the surgical site did not appear normal. This prompted an incisional biopsy of the left mandible. The patient was then referred to our institution for further management.
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      REFERENCES

        • Komp DM.
        Langerhans cell histiocytosis.
        N Engl J Med. 1987; 316: 747-748
        • Lajolo C
        • Campisi G
        • Deli G
        • Littarru C
        • Guiglia R
        • Giuliani M.
        Langerhans's cell histiocytosis in old subjects: two rare case reports and review of the literature.
        Gerodontology. 2012; 29: e1207-e1214
        • Annibali S
        • Cristalli MP
        • Solidani M
        • et al.
        Langerhans cell histiocytosis: oral/periodontal involvement in adult patients.
        Oral Dis. 2009; 15: 596-601
        • Eckardt A
        • Schultze A.
        Maxillofacial manifestations of Langerhans cell histiocytosis: a clinical and therapeutic analysis of 10 patients.
        Oral Oncol. 2003; 39: 687-694
        • Yu Q
        • Wang PZ
        • Shi HM
        • Wang L
        • Sun DX.
        Radiographic findings in Langerhans’ cell disease affecting the mandible.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1995; 79: 251-254
        • Suei Y
        • Tanimoto K
        • Taguchi A
        • Wada T.
        Primary intraosseous carcinoma: review of the literature and diagnostic criteria.
        J Oral Maxillofac Surg. 1994; 52: 580-583
        • Chaisuparat R
        • Coletti D
        • Kolokythas A
        • Ord RA
        • Nikitakis NG.
        Primary intraosseous odontogenic carcinoma arising in an odontogenic cyst or de novo: a clinicopathologic study of six new cases.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006; 101: 194-200
        • Thomas G
        • Pandey M
        • Mathew A
        • et al.
        Primary intraosseous carcinoma of the jaw: pooled analysis of world literature and report of two new cases.
        Int J Oral Maxillofac Surg. 2001; 30: 349-355
        • Klug C
        • Berzaczy D
        • Voracek M
        • Millesi W.
        Preoperative chemoradiotherapy in the management of oral cancer: a review.
        J Craniomaxillofac Surg. 2008; 36: 75-88
        • Hirshberg A
        • Leibovich P
        • Buchner A.
        Metastatic tumors to the jawbones: analysis of 390 cases.
        J Oral Pathol Med. 1994; 23: 337-341
        • Jham BC
        • Salama AR
        • McClure SA
        • Ord RA.
        Metastatic tumors to the oral cavity: a clinical study of 18 cases.
        Head Neck Pathol. 2011; 5: 355-358
        • Thiele OC
        • Freier K
        • Bacon C
        • Flechtenmacher C
        • Scherfler S
        • Seeberger R.
        Craniofacial metastases: a 20-year survey.
        J Craniomaxillofac Surg. 2011; 39: 135-137
        • Hirshberg A
        • Buchner A.
        Metastatic tumours to the oral region. An overview.
        Eur J Cancer B Oral Oncol. 1995; 31B: 355-360
        • Sánchez Aniceto G
        • García Peñín A
        • de la Mata Pages R
        • Montalvo Moreno JJ
        Tumors metastatic to the mandible: analysis of nine cases and review of the literature.
        J Oral Maxillofac Surg. 1990; 48: 246-251
        • Kumar G
        • Manjunatha B.
        Metastatic tumors to the jaws and oral cavity.
        J Oral Maxillofac Pathol. 2013; 17: 71-75
        • Abt NB
        • Lawler ME
        • Zacharias J
        • Lahey ET.
        Primary intraosseous mucoepidermoid carcinoma of the mandible: radiographic evolution and clinicopathological features.
        BMJ Case Rep. 2019; 12e224612
        • Li X
        • Wang F
        • Wang Y
        • Sun S
        • Yang H.
        An unusual case of intraosseous mucoepidermoid carcinoma of the mandible: a case report and literature review.
        Medicine (Baltimore). 2018; 97: e13691
        • He Y
        • Wang J
        • Fu HH
        • Zhang ZY
        • Zhuang QW.
        Intraosseous mucoepidermoid carcinoma of jaws: report of 24 cases.
        Oral Surg Oral Med Oral Pathol Oral Radiol. 2012; 114: 424-429
        • Giridhar P
        • Mallick S
        • Upadhyay AD
        • Rath GK.
        Pattern of care and impact of prognostic factors in the outcome of ameloblastic carcinoma: a systematic review and individual patient data analysis of 199 cases.
        Eur Arch Otorhinolaryngol. 2017; 274: 3803-3810
        • Yoon HJ
        • Hong SP
        • Lee JI
        • Lee SS
        • Hong SD.
        Ameloblastic carcinoma: an analysis of 6 cases with review of the literature.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009; 108: 904-913
        • Miloro M
        • Ghali GE
        • Larsen PE
        • Waite P.
        Peterson's Principles of Oral and Maxillofacial Surgery.
        2nd ed. BC Decker, Hamilton, ON, Canada2004
        • Sood R
        • Gamit M
        • Shah N
        • Mansuri Y
        • Naria G.
        Maxillofacial osteomyelitis in immunocompromised patients: a demographic retrospective study.
        J Maxillofac Oral Surg. 2020; 19: 273-282
        • Baltensperger M
        • Grätz K
        • Bruder E
        • Lebeda R
        • Makek M
        • Eyrich G.
        Is primary chronic osteomyelitis a uniform disease? Proposal of a classification based on a retrospective analysis of patients treated in the past 30 years.
        J Craniomaxillofac Surg. 2004; 32: 43-50
        • Palla B
        • Burian E
        • Fliefel R
        • Otto S.
        Systematic review of oral manifestations related to hyperparathyroidism.
        Clin Oral Investig. 2018; 22: 1-27
        • Brabyn P
        • Capote A
        • Belloti M
        • Zylberberg I.
        Hyperparathyroidism diagnosed due to brown tumors of the jaw: a case report and literature review.
        J Oral Maxillofac Surg. 2017; 75: 2162-2169
        • Silverman Jr, S
        • Ware WH
        • Gillooly Jr, C
        Dental aspects of hyperparathyroidism.
        Oral Surg Oral Med Oral Pathol. 1968; 26: 184-189
        • Martínez-Gavidia EM
        • Bagán JV
        • Milián-Masanet MA
        • Lloria de Miguel E
        • Pérez-Vallés A
        Highly aggressive brown tumour of the maxilla as first manifestation of primary hyperparathyroidism.
        Int J Oral Maxillofac Surg. 2000; 29: 447-449
        • Gorham LW
        • Stout AP.
        Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone): its relation to hemangiomatosis.
        J Bone Joint Surg Am. 1955; 37-A: 985-1004
      1. Romer O. Die Pathologie der Zahne. In: Von Henke F, Lubarsch O., (eds.), Handbuch der speziellen pathologischen Anatomie und Histologie. Vol. 4. Berlin: Springer Verlag; 1924;135-499.

        • Escande C
        • Schouman T
        • Françoise G
        • et al.
        Histological features and management of a mandibular Gorham disease: a case report and review of maxillofacial cases in the literature.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008; 106: e30-e37
        • Chrcanovic BR
        • Gomez RS.
        Gorham-Stout disease with involvement of the jaws: a systematic review.
        Int J Oral Maxillofac Surg. 2019; 48: 1015-1021
        • Nikolaou VS
        • Chytas D
        • Korres D
        • Efstathopoulos N.
        Vanishing bone disease (Gorham-Stout syndrome): a review of a rare entity.
        World J Orthop. 2014; 5: 694-698
        • Hampton J
        • Arthur JF.
        Massive osteolysis affecting the mandible.
        Br Dent J. 1966; 120: 538-541
        • Schiel H
        • Prein J.
        Seven-year follow-up of vanishing bone disease in a 14-year-old girl.
        Head Neck. 1993; 15: 352-356
        • Kato H
        • Ozeki M
        • Fukao T
        • Matsuo M.
        Craniofacial CT findings of Gorham-Stout disease and generalized lymphatic anomaly.
        Neuroradiology. 2016; 58: 801-806
        • Gondivkar SM
        • Gadbail AR.
        Goham-Stout syndrome, a rare clinical entity and review of literature.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2010; 109: e41-e48
        • Paley MD
        • Lloyd CJ
        • Penfold CN.
        Total mandibular reconstruction for massive osteolysis of the mandible (Gorham-Stout syndrome).
        Br J Oral Maxillofac Surg. 2005; 43: 166-168
        • Matsubara M
        • Adachi M
        • Tanuma J
        • Muramatsu Y
        • Sumitomo S.
        Gorham-Stout disease: progressive massive osteolysis of the mandible.
        J Oral Maxillofac Surg Med Pathol. 2016; 28: 147-151
        • Liu M
        • Liu W
        • Qiao C
        • Han B.
        Mandibular Gorham-Stout disease: a case report and literature review.
        Medicine (Baltimore). 2017; 96: e8184
        • Galiay L
        • Simon F
        • Lévy R
        • et al.
        Temporomandibular joint anomalies in pediatric craniofacial Gorham-Stout disease.
        J Craniomaxillofac Surg. 2018; 46: 1179-1184
        • Saify FY
        • Gosavi SR.
        Gorham's disease: a diagnostic challenge.
        J Oral Maxillofac Pathol. 2014; 18: 411-414
        • Johnson PM
        • McClure JG.
        Observations on massive osteolysis; a review of the literature and report of a case.
        Radiology. 1958; 71: 28-42
        • Kim MK
        • Hong JR
        • Kim SG
        • Lee SK.
        Fatal progression of gorham disease: a case report and review of the literature.
        J Oral Maxillofac Surg. 2015; 73: 2352-2360
        • Tie ML
        • Poland GA
        • Rosenow 3rd, EC
        Chylothorax in Gorham's syndrome. A common complication of a rare disease.
        Chest. 1994; 105: 208-213
        • Thompson AA
        • Gorham Patrawala S.
        Stout disease of the mandible, manubrium and cervical spine presenting as bilateral chylothorax.
        BMJ Case Rep. 2021; 14e237638
        • Ferreiro L
        • Suárez-Antelo J
        • Rábade C
        • et al.
        Chylothorax as a debut form of Gorham-Stout disease.
        Pulmonology. 2019; 25: 195-197
        • Wang P
        • Liao W
        • Cao G
        • et al.
        A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report.
        BMC Musculoskelet Disord. 2019; 20: 154
        • Ludwig KF
        • Slone T
        • Cederberg KB
        • Silva AT
        • Dellinger M.
        A new case and review of chylothorax in generalized lymphatic anomaly and Gorham-Stout disease.
        Lymphology. 2016; 49: 73-84
        • Fontanesi J.
        Radiation therapy in the treatment of Gorham disease.
        J Pediatr Hematol Oncol. 2003; 25: 816-817
        • McNeil KD
        • Fong KM
        • Walker QJ
        • Jessup P
        • Zimmerman PV.
        Gorham's syndrome: a usually fatal cause of pleural effusion treated successfully with radiotherapy.
        Thorax. 1996; 51: 1275-1276
        • Duffy BM
        • Manon R
        • Patel RR
        • Welsh JS
        A case of Gorham's disease with chylothorax treated curatively with radiation therapy.
        Clin Med Res. 2005; 3: 83-86
        • Hagberg H
        • Lamberg K
        • Aström G.
        Alpha-2b interferon and oral clodronate for Gorham's disease.
        Lancet. 1997; 350: 1822-1823
        • Barili F
        • Polvani G
        • Topkara VK
        • et al.
        Administration of octreotide for management of postoperative high-flow chylothorax.
        Ann Vasc Surg. 2007; 21: 90-92
        • Sharkey AJ
        • Rao JN.
        The successful use of octreotide in the treatment of traumatic chylothorax.
        Tex Heart Inst J. 2012; 39: 428-430
        • Ulíbarri JI
        • Sanz Y
        • Fuentes C
        • Mancha A
        • Aramendia M
        • Sánchez S.
        Reduction of lymphorrhagia from ruptured thoracic duct by somatostatin.
        Lancet. 1990; 336: 258
        • Jain A
        • Singh SN
        • Singhal P
        • Sharma MP
        • Grover M.
        A prospective study on the role of octreotide in management of chyle fistula neck.
        Laryngoscope. 2015; 125: 1624-1627
        • Nussenbaum B
        • Liu JH
        • Sinard RJ.
        Systematic management of chyle fistula: the Southwestern experience and review of the literature.
        Otolaryngol Head Neck Surg. 2000; 122: 31-38