Permanent central diabetes insipidus as a complication of sphenoid sinus mucocele




Abstract


Although mucocele is a benign lesion, its unavoidable expansions may result in irreversible damages in adjacent organs. In spheno-ethmoid mucoceles which are extremely rare, this condition may cause more severe problems. Central diabetes insipidus, developed secondary to sphenoid sinus mucocele, was detected in a 54-year-old male patient, who underwent endoscopic sinus surgery 2 times due to nasal polyposis. Endoscopic sphenoid mucocele marsupialization was performed to the patient, but despite partial regression in the 1-year follow up, complete recovery was not observed.



Introduction


Generally mucoceles are determined in frontal and ethmoid sinuses but they are extremely rare in sphenoid sinus. Since sphenoid mucoceles grow slowly, they are only symptomatic when they yield large masses. These benign but local invasive masses, due to their expansions to the contiguous tissues, cause different symptoms. If a definitive laboratory analysis is not achieved, they are easily misdiagnosed and mistreated. In the etiology of the mucocele, other than the inflammatory processes including sinusitis or nasal polyposis, the iatrogenic causes such as endoscopic sinus surgery are also present . Though they are rare entities, iatrogenic sphenoid sinus pathologies may lead to severe complications due to the critical anatomical surroundings of the sphenoid sinus.


In this paper, a sphenoid mucocele case diagnosed during diabetes insipidus (DI) evaluation was discussed with the literature.





Case presentation


A 54-year-old male patient, was admitted to the endocrinology department with excessive water consumption and thamuria for 6–7 months. His laboratory evaluation with the pre-diagnosis of DI revealed a serum sodium of 144 mEq/L (132–146), serum potassium of 4.1 mEq/L (3.5–5.5) and a urinary density of 1003 g/cm 3 . For the exact diagnosis of patient, water restriction–antidiuretic hormone (ADH) test was performed and at eighth hour of the test following ADH administration, a more than 50% increase in urinary osmolarity was observed (before ADH: 331 mosm/kg, after ADH: 556 mosm/kg). The patient was diagnosed with central DI and intranasal desmopressin treatment of 10 mcg 1 × 1 was given. His adenohypophyseal hormones were normal. After this treatment, water consumption and thamuria were fully regressed. The patient had no other endocrine dysfunctions except central DI. In his history there was no head trauma, radiotherapy or intracranial diseases but it was learnt that he had endoscopic sinus surgery 2 times (6 and 2 years ago) due to nasal polyps. In magnetic resonance imaging (MRI) performed to determine the etiology of central DI, a lesion with approximately 5 × 3 cm dimensions, fulfilling sphenoid sinus and expanding through sellar region and nasal cavity was reported. This lesion was hypointense in T1 weighed scans but hyperintense in T2 weighed scans. Posterior of the lesion a relatively thin hypophysis tissue was observed ( Fig. 1 ). Paranasal sinus computed tomography revealed an appearance compatible with sinonasal polyposis together with a defect in sellar base. Afterward, the patient was consulted to our clinic. In his nasal endoscopic examination, there was no middle concha and polypoid nasal mucosa was observed in the middle meatus. Other otorhinolaryngologic examinations were normal. We performed transnasal endoscopic sinus surgery with sphenoid sinus mucocele marsupialization. After the marsupialization, it was observed that sellar floor was eroded markedly due to mucocele in sphenoid sinus. Operation was completed successfully and there were no complications observed in intra and postoperative periods. In the third month after operation, desmopressin treatment was interrupted for a while and the patient was reevaluated for DI. Although his symptoms and signs were better than the preoperative period, desmopressin had to be started again. Despite a regression in the patient’s central DI, complete recovery was not observed in the clinical picture in the 1 year follow up postoperatively. Currently, the patient still receives oral desmopressin treatment regularly.




Fig. 1


MRI images of lesion. Images showed that the lesion fulfilled sphenoid sinus and expanded through sellar region and nasal cavity. This lesion was hypointense in T1 weighed scans (A) but hyperintense in T2 weighed scans (B). A; anterior side, R: right side.





Case presentation


A 54-year-old male patient, was admitted to the endocrinology department with excessive water consumption and thamuria for 6–7 months. His laboratory evaluation with the pre-diagnosis of DI revealed a serum sodium of 144 mEq/L (132–146), serum potassium of 4.1 mEq/L (3.5–5.5) and a urinary density of 1003 g/cm 3 . For the exact diagnosis of patient, water restriction–antidiuretic hormone (ADH) test was performed and at eighth hour of the test following ADH administration, a more than 50% increase in urinary osmolarity was observed (before ADH: 331 mosm/kg, after ADH: 556 mosm/kg). The patient was diagnosed with central DI and intranasal desmopressin treatment of 10 mcg 1 × 1 was given. His adenohypophyseal hormones were normal. After this treatment, water consumption and thamuria were fully regressed. The patient had no other endocrine dysfunctions except central DI. In his history there was no head trauma, radiotherapy or intracranial diseases but it was learnt that he had endoscopic sinus surgery 2 times (6 and 2 years ago) due to nasal polyps. In magnetic resonance imaging (MRI) performed to determine the etiology of central DI, a lesion with approximately 5 × 3 cm dimensions, fulfilling sphenoid sinus and expanding through sellar region and nasal cavity was reported. This lesion was hypointense in T1 weighed scans but hyperintense in T2 weighed scans. Posterior of the lesion a relatively thin hypophysis tissue was observed ( Fig. 1 ). Paranasal sinus computed tomography revealed an appearance compatible with sinonasal polyposis together with a defect in sellar base. Afterward, the patient was consulted to our clinic. In his nasal endoscopic examination, there was no middle concha and polypoid nasal mucosa was observed in the middle meatus. Other otorhinolaryngologic examinations were normal. We performed transnasal endoscopic sinus surgery with sphenoid sinus mucocele marsupialization. After the marsupialization, it was observed that sellar floor was eroded markedly due to mucocele in sphenoid sinus. Operation was completed successfully and there were no complications observed in intra and postoperative periods. In the third month after operation, desmopressin treatment was interrupted for a while and the patient was reevaluated for DI. Although his symptoms and signs were better than the preoperative period, desmopressin had to be started again. Despite a regression in the patient’s central DI, complete recovery was not observed in the clinical picture in the 1 year follow up postoperatively. Currently, the patient still receives oral desmopressin treatment regularly.


Aug 24, 2017 | Posted by in OTOLARYNGOLOGY | Comments Off on Permanent central diabetes insipidus as a complication of sphenoid sinus mucocele
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