GH-secreting pituitary macroadenoma (acromegaly) associated with progressive dental malocclusion and refractory CPAP treatment

Abstract

BackgroundA link between progressive dental malocclusion, the use of a continuous positive airway pressure mask and GH-secreting pituitary macroadenoma (acromegaly) has not been previously reported. The present clinicopathological analysis stresses that tooth malposition should not be seen exclusively as a local process.Case presentationA 62-year-old caucasian man with no relevant medical history reported difficulty chewing food and perceived voice alteration during his annual periodontal check-up. He also referred stiffness of the tongue, face, and submandibular area. The patient had been diagnosed with obstructive sleep apnea syndrome two years previously, since when he had worn a continuous positive airway pressure device during sleep. Exploration of the occlusion revealed significant changes: an atypical left lateral and anterior open bite with major buccoversion of teeth 33, 34, 35, 36. Inspection of the soft tissue revealed only macroglossia, although external palpation indicated a subcutaneous stiffness of the submandibular area. General analytical tests, including hormone profiles, and magnetic resonance imaging confirmed the diagnosis of acromegaly induced by a pituitary adenoma. Intrasellar tumor resection via transsphenoidal approach was performed. After surgery, the patient already noted a marked improvement of all symptoms associated with the acromegaly. Desaturation data also evolved favourably and the pulmonologist advised the patient to abandon the continuous positive airway pressure treatment.ConclusionProgressive dental malocclusion may be associated with a systemic disease and the use of a nasal mask with premaxillary support may distort the diagnosis of acromegaly.

The implications of endocrinology in orthodontics: Literature review

“…An excessive production of growth hormone after adolescence causes acromegaly 6,7 . The characteristic extra-oral features include a prominent forehead, growth of the nose and ears, deepening of the nasolabial folds and mandibular prognathism 8,9 .…”

Practical Aspects of Interface Application in CPAP Treatment

“…It is important to note that the existence of progressive dental malocclusion (i.e. mandibular protrusion and open bite) in nasal mask users may also be associated with a very rare systemic disease (such as acromegaly) that should be excluded [10]. Compared to CPAP therapy, oral-appliance therapy for sleep apnea has a slightly higher risk for craniofacial or dental changes.…”

Association of total, acylated and unacylated ghrelin with apolipoprotein A1 and insulin concentrations in acromegalic patients

“…9 First line treatment is transsphenoidal surgery, sometimes preceded by long-acting somatostatin analogs (SSAs) administration. 10 While in 70% of cases acromegaly is associated with pituitary macroadenoma, 11 most patients require additional treatment. For those with persistent disease following surgery, pharmacological treatment or/and radiotherapy is recommended.…”