- Email: [email protected]
Acromegaly in an orthodontic patient
CLINICIAN’S CORNER
Acromegaly in an orthodontic patient Robert Willer Farinazzo Vitral,a Orlando Motohiro Tanaka,b Marcelo Reis Fraga,c and Edvaldo Antônio Ribeiro Rosad Juiz de Fora and Curitiba, Brazil A 32-year-old white man presented for orthodontic treatment with the chief complaint of mandibular prognathism; he was later found to have acromegaly. General information about pituitary adenomas, specifically growth hormone adenomas, is given, and treatment options are discussed. (Am J Orthod Dentofacial Orthop 2006;130:388-90)
P
ituitary adenomas are a diverse group of benign neoplasms1,2 and the most commonly encountered intracranial tumor.2 They have been found in 10% of the normal population by using magnetic resonance imaging and in up to 25% of persons undergoing autopsies.3-5 Pituitary adenomas have various clinical manifestations that are related to excessive hormone secretion by the tumor, hormone deficits by the normal pituitary gland, and expansion of the tumor mass.2 Most adenomas are considered benign.6 Pituitary adenomas are broadly classified according to either size or functional status. Adenomas measuring up to 10 mm are considered microadenomas and are usually located in sella turcica; those larger than 10 mm are termed macroadenomas and can be totally intrasellar but are often associated with extrasellar extension.1,7 Another classification scheme distinguishes whether the tumors are associated with clinical and laboratory evidence of excess hormone secretion. The traditional nomenclature describes adenomas as hyperfunctioning or nonfunctioning. The 3 most common hyperfunctioning adenomas are the prolactin-secreting adenoma, the growth-hormone (GH) adenoma, and the corticotropin adenoma.1 GH-secreting adenomas account for approximately 20% of all pituitary tumors. In adults, they manifest clinically as acromegaly and gigantism if the onset of the disease is before the closure of the epiphyseal plates.1,2,8 Patients with acromegaly experience proa
Associate professor and chair, Universidade Federal de Juiz de Fora, Juiz de Fora, Brazil. b Senior professor, Pontifícia Universidade Católica, Curitiba, Brazil. c Professor, Universidade Federal de Juiz de Fora, Juiz de Fora, Brazil. d Associate professor, Pontifícia Universidade Católica do Paraná, Curitiba, Brazil. Reprint requests to: Robert Willer Farinazzo Vitral, Av Rio Branco 2595/1604, Juiz de Fora, Minas Gerais, Brazil, CEP 36010 907; e-mail, robertvitral@ acessa.com. Submitted, September 2004; revised and accepted, November 2004. 0889-5406/$32.00 Copyright © 2006 by the American Association of Orthodontists. doi:10.1016/j.ajodo.2004.11.038
388
gressive and often insidious cosmetic changes, including frontal bossing, prognathism, macroglossia, and increased hand and foot sizes.1 Patients also experience cardiovascular hypertension, congestive heart failure, and impaired glucose metabolism. Tissue hypertrophy also increases the incidence of diabetes mellitus, sleep apnea, lumbar stenosis, and carpal tunnel syndrome.1,9,10 GH excess in adolescents causes the additional features of excessive long bone growth and subsequent increased stature.1 Most patients have a 5- to 10-year history of alterations in features, bone overgrowth, soft-tissue swelling, and skin changes.11 Diagnosis of acromegaly is relatively simple and depends on clinical recognition of the signs and symptoms. Measurement of GH response to a glucose load is the standard diagnostic test.12 Magnetic resonance imaging (MRI) has emerged as the imaging modality of choice for evaluating the pituitary gland.1 Superb resolution allows small lesions to be visualized and clearly defines the relationship of larger lesions to adjacent structures.5 REPORT OF A CASE
A 32-year-old white man presented for orthodontic treatment, concerned with diastemas in his mandibular arch that were related to periodontally compromised teeth and mandibular prognathism. His dental history included periodontal treatment because of generalized horizontal bone loss, endodontic treatment of the maxillary right second premolar and the mandibular right first molar, and loss of the maxillary right first molar, which was replaced prosthetically. He had a Class III malocclusion with a concave profile, posterior and anterior crossbites, and diastemas in the anteroinferior segment (Fig 1). Periapical radiographs showed slight apical root resorption in the mandibular molars and the maxillary incisors, and the cephalometric analysis (Fig 2) indicated a Class III skeletal discrepancy (SNA angle ⫽ 77°, SNB angle ⫽ 84°, ANB angle ⫽ ⫺7°).
Vitral et al 389
American Journal of Orthodontics and Dentofacial Orthopedics Volume 130, Number 3
Fig 1. Intraoral photographs.
Fig 2. Lateral cephalometric radiograph and cephalometric tracing.
Fig 3. MRIs of tumor site: A, before surgery; B, 1 year after surgery.
The patient reported that his mandibular prognathism began to develop 4 years previously, and he mentioned 2 other facts: his shoe size had increased considerably during that time period, and his wedding ring no longer fit.
DISCUSSION
Based on the patient’s signs and symptoms, the diagnosis of acromegaly was considered. Mandibular prognathism is among the most commonly found oral manifestations of acromegaly, and it was the main
390 Vitral et al
reason that this patient sought orthodontic treatment. Because of macroglossia, his tongue was enlarged and had became more furrowed. It is likely that the diastemas in the mandibular arch were also partially related to the enlarged tongue. Although generalized apical root resorption is a frequent finding in patients with acromegaly,8 only the mandibular molars and the maxillary incisors showed such resorption. Frontal bossing, prognathism, macroglossia, and increased hand and foot sizes are often described in the literature as cosmetic changes associated with GH adenomas,1 and all were present in this patient. Enlarged facial features and excessive acral growth are found in nearly all subjects with acromegaly.5 Other symptoms this patient reported included fatigue, daytime somnolence, and joint pain. Decreased energy, osteoarthritis, and somnolence occur in about 50% of patients.5 The patient was referred for a medical examination, and the diagnosis of pituitary macroadenoma was confirmed. Available treatment options include surgery, medication, and radiation therapy.2 Trans-sphenoidal surgery is regarded as the first-line treatment of acromegaly.5 Long-term remission is achieved in 50%5 to 67%13 of patients, but safely removing all tumor tissue can be difficult. Regardless of the initial therapy choice, follow-up of these patients should continue indefinitely.2 This patient was treated with trans-sphenoidal surgery. Figure 3, A, shows an MRI scan of the tumor before surgery; 1 year after sugery, a new MRI, Figure 3, B, shows the volumetric reduction of the gland. Today, the patient is taking medication, and orthodontic
American Journal of Orthodontics and Dentofacial Orthopedics September 2006
treatment has been postponed until he is cleared medically. REFERENCES 1. Jane JR Jr, Laws ER Jr. Surgical management of pituitary adenomas. Singapore Med J 2002;6:318-23. 2. Arafah BM, Nasrallah MP. Pituitary tumors: pathophysiology, clinical manifestations and management. Endocr Relat Cancer 2001;8:287-305. 3. Hall WA, Luciano MG, Doppman JL, Patronas NJ, Oldfield EH. Pituitary magnetic resonance imaging in normal human volunteers: occult adenomas in the general population. Ann Intern Med 1994;120:817-20. 4. Burrow GN, Wortzman G, Rewcastle NB, Holgate RC, Kovacs K. Microadenomas of the pituitary and abnormal sellar tomograms in an unselected autopsy series. N Engl J Med 1981;304: 156-8. 5. Hurley DM, Ho KKY. Pituitary disease in adults. Med J Aust 2004;180:419-25. 6. Lubke D, Saeger W. Carcinomas of the pituitary: definition and revision of the literature. Gen Diagn Pathol 1995;141:81-92. 7. Gsponer J, DeTribolet N, Deruaz JP, Janzer R, Uske A, Mirimanoff RO, et al. Diagnosis, treatment, and outcome of pituitary tumors and other abnormal intrasellar masses. Retrospective analysis of 353 patients. Medicine 1999;78:236-69. 8. Shafer WG, Hine MK, Levy BM. A textbook of oral pathology. Philadelphia: W. B. Saunders; 1974. 9. Melmed S. Acromegaly. N Engl J Med 1990;322:966-77. 10. Klibansk A, Zervas NT. Diagnosis and management of hormonesecreting pituitary adenomas. N Engl J Med 1991;324:822-31. 11. Furman K, Ezzat S. Psychological features of acromegaly. Psychother psychosom 1998;67:147-53. 12. Arafah BM, Rosenzweig JF, Fenstermaker R, Salazar R, McBride CE, Selman WR. Value of growth hormone dynamics and somatomedin—C levels in predicting the long term benefit after transsphenoidal surgery for acromegaly. J Lab Clin Med 1987;109:346-64. 13. Laws ER, Vance ML, Thapar K. Pituitary surgery for the management of acromegaly. Horm Res 2000;53:71-5.
Acromegaly in an orthodontic patient Robert Willer Farinazzo Vitral,a Orlando Motohiro Tanaka,b Marcelo Reis Fraga,c and Edvaldo Antônio Ribeiro Rosad Juiz de Fora and Curitiba, Brazil A 32-year-old white man presented for orthodontic treatment with the chief complaint of mandibular prognathism; he was later found to have acromegaly. General information about pituitary adenomas, specifically growth hormone adenomas, is given, and treatment options are discussed. (Am J Orthod Dentofacial Orthop 2006;130:388-90)
P
ituitary adenomas are a diverse group of benign neoplasms1,2 and the most commonly encountered intracranial tumor.2 They have been found in 10% of the normal population by using magnetic resonance imaging and in up to 25% of persons undergoing autopsies.3-5 Pituitary adenomas have various clinical manifestations that are related to excessive hormone secretion by the tumor, hormone deficits by the normal pituitary gland, and expansion of the tumor mass.2 Most adenomas are considered benign.6 Pituitary adenomas are broadly classified according to either size or functional status. Adenomas measuring up to 10 mm are considered microadenomas and are usually located in sella turcica; those larger than 10 mm are termed macroadenomas and can be totally intrasellar but are often associated with extrasellar extension.1,7 Another classification scheme distinguishes whether the tumors are associated with clinical and laboratory evidence of excess hormone secretion. The traditional nomenclature describes adenomas as hyperfunctioning or nonfunctioning. The 3 most common hyperfunctioning adenomas are the prolactin-secreting adenoma, the growth-hormone (GH) adenoma, and the corticotropin adenoma.1 GH-secreting adenomas account for approximately 20% of all pituitary tumors. In adults, they manifest clinically as acromegaly and gigantism if the onset of the disease is before the closure of the epiphyseal plates.1,2,8 Patients with acromegaly experience proa
Associate professor and chair, Universidade Federal de Juiz de Fora, Juiz de Fora, Brazil. b Senior professor, Pontifícia Universidade Católica, Curitiba, Brazil. c Professor, Universidade Federal de Juiz de Fora, Juiz de Fora, Brazil. d Associate professor, Pontifícia Universidade Católica do Paraná, Curitiba, Brazil. Reprint requests to: Robert Willer Farinazzo Vitral, Av Rio Branco 2595/1604, Juiz de Fora, Minas Gerais, Brazil, CEP 36010 907; e-mail, robertvitral@ acessa.com. Submitted, September 2004; revised and accepted, November 2004. 0889-5406/$32.00 Copyright © 2006 by the American Association of Orthodontists. doi:10.1016/j.ajodo.2004.11.038
388
gressive and often insidious cosmetic changes, including frontal bossing, prognathism, macroglossia, and increased hand and foot sizes.1 Patients also experience cardiovascular hypertension, congestive heart failure, and impaired glucose metabolism. Tissue hypertrophy also increases the incidence of diabetes mellitus, sleep apnea, lumbar stenosis, and carpal tunnel syndrome.1,9,10 GH excess in adolescents causes the additional features of excessive long bone growth and subsequent increased stature.1 Most patients have a 5- to 10-year history of alterations in features, bone overgrowth, soft-tissue swelling, and skin changes.11 Diagnosis of acromegaly is relatively simple and depends on clinical recognition of the signs and symptoms. Measurement of GH response to a glucose load is the standard diagnostic test.12 Magnetic resonance imaging (MRI) has emerged as the imaging modality of choice for evaluating the pituitary gland.1 Superb resolution allows small lesions to be visualized and clearly defines the relationship of larger lesions to adjacent structures.5 REPORT OF A CASE
A 32-year-old white man presented for orthodontic treatment, concerned with diastemas in his mandibular arch that were related to periodontally compromised teeth and mandibular prognathism. His dental history included periodontal treatment because of generalized horizontal bone loss, endodontic treatment of the maxillary right second premolar and the mandibular right first molar, and loss of the maxillary right first molar, which was replaced prosthetically. He had a Class III malocclusion with a concave profile, posterior and anterior crossbites, and diastemas in the anteroinferior segment (Fig 1). Periapical radiographs showed slight apical root resorption in the mandibular molars and the maxillary incisors, and the cephalometric analysis (Fig 2) indicated a Class III skeletal discrepancy (SNA angle ⫽ 77°, SNB angle ⫽ 84°, ANB angle ⫽ ⫺7°).
Vitral et al 389
American Journal of Orthodontics and Dentofacial Orthopedics Volume 130, Number 3
Fig 1. Intraoral photographs.
Fig 2. Lateral cephalometric radiograph and cephalometric tracing.
Fig 3. MRIs of tumor site: A, before surgery; B, 1 year after surgery.
The patient reported that his mandibular prognathism began to develop 4 years previously, and he mentioned 2 other facts: his shoe size had increased considerably during that time period, and his wedding ring no longer fit.
DISCUSSION
Based on the patient’s signs and symptoms, the diagnosis of acromegaly was considered. Mandibular prognathism is among the most commonly found oral manifestations of acromegaly, and it was the main
390 Vitral et al
reason that this patient sought orthodontic treatment. Because of macroglossia, his tongue was enlarged and had became more furrowed. It is likely that the diastemas in the mandibular arch were also partially related to the enlarged tongue. Although generalized apical root resorption is a frequent finding in patients with acromegaly,8 only the mandibular molars and the maxillary incisors showed such resorption. Frontal bossing, prognathism, macroglossia, and increased hand and foot sizes are often described in the literature as cosmetic changes associated with GH adenomas,1 and all were present in this patient. Enlarged facial features and excessive acral growth are found in nearly all subjects with acromegaly.5 Other symptoms this patient reported included fatigue, daytime somnolence, and joint pain. Decreased energy, osteoarthritis, and somnolence occur in about 50% of patients.5 The patient was referred for a medical examination, and the diagnosis of pituitary macroadenoma was confirmed. Available treatment options include surgery, medication, and radiation therapy.2 Trans-sphenoidal surgery is regarded as the first-line treatment of acromegaly.5 Long-term remission is achieved in 50%5 to 67%13 of patients, but safely removing all tumor tissue can be difficult. Regardless of the initial therapy choice, follow-up of these patients should continue indefinitely.2 This patient was treated with trans-sphenoidal surgery. Figure 3, A, shows an MRI scan of the tumor before surgery; 1 year after sugery, a new MRI, Figure 3, B, shows the volumetric reduction of the gland. Today, the patient is taking medication, and orthodontic
American Journal of Orthodontics and Dentofacial Orthopedics September 2006
treatment has been postponed until he is cleared medically. REFERENCES 1. Jane JR Jr, Laws ER Jr. Surgical management of pituitary adenomas. Singapore Med J 2002;6:318-23. 2. Arafah BM, Nasrallah MP. Pituitary tumors: pathophysiology, clinical manifestations and management. Endocr Relat Cancer 2001;8:287-305. 3. Hall WA, Luciano MG, Doppman JL, Patronas NJ, Oldfield EH. Pituitary magnetic resonance imaging in normal human volunteers: occult adenomas in the general population. Ann Intern Med 1994;120:817-20. 4. Burrow GN, Wortzman G, Rewcastle NB, Holgate RC, Kovacs K. Microadenomas of the pituitary and abnormal sellar tomograms in an unselected autopsy series. N Engl J Med 1981;304: 156-8. 5. Hurley DM, Ho KKY. Pituitary disease in adults. Med J Aust 2004;180:419-25. 6. Lubke D, Saeger W. Carcinomas of the pituitary: definition and revision of the literature. Gen Diagn Pathol 1995;141:81-92. 7. Gsponer J, DeTribolet N, Deruaz JP, Janzer R, Uske A, Mirimanoff RO, et al. Diagnosis, treatment, and outcome of pituitary tumors and other abnormal intrasellar masses. Retrospective analysis of 353 patients. Medicine 1999;78:236-69. 8. Shafer WG, Hine MK, Levy BM. A textbook of oral pathology. Philadelphia: W. B. Saunders; 1974. 9. Melmed S. Acromegaly. N Engl J Med 1990;322:966-77. 10. Klibansk A, Zervas NT. Diagnosis and management of hormonesecreting pituitary adenomas. N Engl J Med 1991;324:822-31. 11. Furman K, Ezzat S. Psychological features of acromegaly. Psychother psychosom 1998;67:147-53. 12. Arafah BM, Rosenzweig JF, Fenstermaker R, Salazar R, McBride CE, Selman WR. Value of growth hormone dynamics and somatomedin—C levels in predicting the long term benefit after transsphenoidal surgery for acromegaly. J Lab Clin Med 1987;109:346-64. 13. Laws ER, Vance ML, Thapar K. Pituitary surgery for the management of acromegaly. Horm Res 2000;53:71-5.