Abstract
PurposeSalivary gland (SG) tissue and derived neoplasms may occur in the sellar region. As the current literature is mostly limited to case reports, the puzzling case of an inflammatory SG removed by transsphenoidal surgery (TS) and mimicking a prolactinoma prompted us to perform the first systematic review of these unusual conditions.MethodsA systematic literature search was conducted according to the PRISMA guidelines. Forty-four individual cases—non-neoplastic enlarged salivary glands (NNESG, n = 15), primary benign (n = 7) and malignant (n = 8) ectopic salivary tumours (ST) and sellar metastasis from eutopic primary ST (n = 14)—were suitable for the analysis of clinical, radiological and pathological characteristics. Therapeutic outcome was reviewed as a secondary endpoint.ResultsAll cases were diagnosed after surgery. NNESG commonly affected young and/or female patients, typically leading to headaches and hyperprolactinemia and originating close to the neurohypophysis. Submucosal SG should be excluded before concluding to an intrasellar NNESG after TS. No gender or age predominance was found for primary ectopic ST, which present as large tumors, with histological phenotypes similar to common ST. Hypopituitarism and diabetes insipidus were more frequent in ST than in NNESG. NNESG and benign ectopic ST rarely recur. Malignant ectopic ST should be distinguished from secondary localizations of eutopic ST reaching the sella by contiguity or metastatic spread; both share a frequent unfavorable outcome.ConclusionSellar neoplasms derived from SG are rare but misleading conditions and pituitary dysfunction is likely to be more common than currently reported. Appropriate pathological evaluation and multidisciplinary approach are required.
Highlights
Ectopic salivary gland (SG) tissue may occur in different sites of the body: extra-cranially [1–6] and intra-cranially, Sapienza”, Rome, RM, Italy 3 Department of Radiological, Oncological and PathologicalSciences, University “La Sapienza”, Rome, RM, Italy 4 Department of Neurology and Psychiatry, University “LaSapienza”, Rome, RM, Italy 5 Department of Biotechnological and Applied ClinicalSciences, University of L’Aquila, Via Vetoio, Coppito 2, 67100 L’Aquila, AQ, Italy with sellar and extra-sellar localizations (e.g.: optic nerve sheath, cerebellopontine angle) [7–9]
Because PitNETs were originally reported as pituitary adenomas (PA) in all papers, we elected to maintain this terminology to report the pre-operative diagnosis
The heterogeneity of radiological descriptions, in part reflecting a variety of pathological histotypes [51], confirms the lack of strongly suggestive features, cystic components of variable protein content were frequent in NNESG
Summary
Ectopic salivary gland (SG) tissue may occur in different sites of the body: extra-cranially (larynx, gastrointestinal tract, middle ear, chest wall) [1–6] and intra-cranially, Sapienza”, Rome, RM, Italy 3 Department of Radiological, Oncological and Pathological. University of L’Aquila, Via Vetoio, Coppito 2, 67100 L’Aquila, AQ, Italy with sellar and extra-sellar localizations (e.g.: optic nerve sheath, cerebellopontine angle) [7–9]. Symptomatic enlargement of ectopic SG rests may be non-neoplastic (NNESG) or due to benign or malignant salivary tumours (ST) that mimic other non-functioning lesions, and the diagnosis relies on pathology where surgery is indicated. Because malignant ST derived from major or minor eutopic SG may reach the sella through local invasion or blood spread [21–23], an extra-sellar origin should be excluded before concluding to a primary ectopic SG malignancy [24].
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