Volume 1, Issue 2 , Pages 119-122, June 2006
Nasopharyngeal cyst after adenoidectomy in a child with liver transplantation
Article Outline
Summary
We report a very unusual case of a liver transplanted child referred to our Department under clinical suspicion of malignancy. The patient underwent an adenotonsillectomy under general anesthesia without perioperative problems. The post-operative pathological diagnosis was follicular hyperplasia.
Six months later, the patient had a progressive impairment of his breathing. Oropharyngeal examination and magnetic resonance imaging showed a cyst mass hanging behind the soft palate. The patient was scheduled for excision-biopsy of the cystic mass, which was performed without complications. Pathologic diagnosis of the tissue removed in this second operation was granulation tissue. The child is alive and well 5 years after the last surgical procedure.
Keywords: Lymphoproliferative disease, Adenoidectomy, Liver transplant, Complications, Unusual
1. Introduction
Organ transplantation in pediatric patients is one of the more important medical advances in recent years. Since cases of different organ transplants have increased in children, the need to perform operations on the tonsils and/or adenoids (T&A) of post-transplanted patients is increasing [1], [2].
In pediatric T&A bleeding remains as the most important complication that shortly occurs after the procedure [3]. Other common complications associated with these procedures include airway compromise, acute otitis media, velopalatine insufficiency, and torticollis [4]. All of them comprise an overall complication rate of 1.4% [5]. Nevertheless, a wide variety of unusual complications can be found in the literature, some of them of an extremely serious nature [6].
This article presents an unusual case of nasopharyngeal cyst that occurs after adenoidectomy in a pediatric liver transplanted patient.
2. Case report
A 3-years-old male patient was referred to our Department under clinical suspicion of malignancy because of the progressive enlargement of the tonsils in a short period of time. He received a liver transplant 3 months before. The child did not have weight loss. There was no history of recent infection, head and neck trauma, or thyrotoxicosis. Physical examination revealed obstructive hyperplasia of tonsils (grade IV/IV) and adenoids (Fig. 1). No neck nodes were palpated. The patient underwent a T&A under general anesthesia without perioperative problems. Adenoidectomy was performed by curettage of the nasopharynx (electrical and/or chemical cautery, or any suture material were not employed during adenoidectomy), and tonsillectomy by “classical” dissection with pillar suture. After curettage of the adenoidal tissue, gauze packing was left 10
min. The post-operative pathological diagnosis was follicular hyperplasia (Fig. 2).
Fig. 1.
Preoperative X-ray film of the lateral aspect of the neck showing hyperplastic adenoids.
Fig. 2.
Follicular hyperplasia. There is great range in the size of follicles, some being very large. Note the rim of dark-stained mantle zone lymphocytes surrounding the active germinal centers (H&E, 40×).
Six months later, the patient had a progressive impairment of his breathing. A new oropharyngeal examination was performed showing a cystic mass hanging behind the soft palate. Magnetic resonance imaging (MRI) showed a cystic mass depending of the nasopharynx (Fig. 3). The patient was scheduled for excision-biopsy of the mass which was performed without complications. Pathologic diagnosis of the tissue removed in this second operation was granulation tissue (Fig. 4). Although the blood samples were positive before and after operation to Epstein-Barr virus (EBV) and lymphotropic herpes simplex virus, tissue samples of T&A specimens were negative for EBV. The child is alive and well 5 years after the last surgical procedure.
Fig. 3.
Magnetic resonance imaging 6 months after the initial surgical procedure. Note the presence of a cystic mass in the nasopharynx.
Fig. 4.
Granulation tissue. Note the presence of numerous vessels embedded in a loose connective tissue matrix containing mesenchimal cells and occasional inflammatory cells (H&E, 100×).
3. Discussion
The utility of T&A in organ transplanted children is very broad and include biopsy to exclude post-transplant lymphoproliferative disease (PTLD) in Waldeyer's ring, erradication of episodes of recurrent tonsillitis, and treatment of persistent EBV in the adenotonsillar area [2], [7]. Complications following T&A procedures can be clasiffied based on the timing of their occurrence in relation to surgery, namely: intraoperative, immediate post-operative (<24h), delayed (<2 weeks), and long-term [8]. Although common and uncommon complications can be seen in the intraoperative, immediate and delayed periods, in the long-term group there are only rare complications.
Intraoperative complications encompass mainly bleeding, airway obstruction, and anesthetic problems [4], [9]. Atypical forms of haemorrhage can arise if a vascular damage of aberrant head and neck vessels occurs [6], [10], [11], [12], [13]. Likewise, unusual cases of respiratory obstruction may be seen associated with insertion of post-nasal pack [6]. Complications linked with general anesthesia include cardiac arrest, failure to intubate and malignant hypertermia [5], [9]. On the other hand, surgical trauma affecting the tip of the tongue, tonsillar pillars, soft palate, uvula and pharyngeal walls have been ocasionally reported in the medical literature [14]. Breakage of instruments rarely complicates tonsil and adenoid removal being the accident seldom serious [6]. Finally, intraoperative oral cavity fire due to electrocautery malfunction has been also described [9].
Haemorrhage, breathing impairment, infection, severe nausea and dehydration, may be present in the immediate post-operative period [9], [14]. In the same manner, subcutaneous emphysema, air embolism, Grisel's syndrome and Eagle's syndrome, bilateral visual loss (associated with local infiltration of the tonsillar bed) and paroxysmal myoglobinuria have been seldom described at this time [9], [14], [15], [16]. An unusual vascular complication most specifically linked with transplanted patients is hepatic artery thrombosis observed when excessive perioperative circulation changes arise [1], [2].
Classically, delayed complications include bleeding, otalgia, acute otitis media, velopalatine insufficiency and torticollis [4]. Most rarely sagittal sinus thrombosis from severe dehydration, paralysis of the vagus and hypoglossal nerve, and meningitis and/or brain abscess, may also occur at this moment after the procedure [6], [9], [14].
Finally, long-term complications as severe haemorrhage, external carotid pseudoaneurysms or arterial dissection can be seen after T&A [17], [18], [19]. Ectopic salivation of minor salivary glands into the tonsil fossae has been observed [14]. The nasopharyngeal cyst developed by our patient may, perhaps, occur due to two possible mechanisms: (a) post-surgical obstruction of a nasopharyngeal minor salivary gland duct after the first adenoid curettage; and (b) unsuspected existence of a cyst derived from the embryonal pharyngeal bursa [20]. This formation is separated from the nasopharyngeal mucosa by a membrane or by the fibers of the longus capitis muscle and hence has not been removed at the previous adenoidectomy [20]. Likewise, conversion disorders after T&A procedures have been also described in this period.
4. Conclusion
There is little information in the medical literature about rare complications after T&A. A very unusual case of nasopharyngeal cyst after T&A in a liver transplanted patient is presented. This clinical problem underscores the importance of expecting unusual problems in this type of surgical procedures.
References
- . Posttransplant lymphoproliferative disease presenting as sudden respiratory arrest in a three-year-old child. Ann. Otol. Rhinol. Laryngol. 1997;106:244–247
- . Tonsillectomy in a boy liver transplant. Nippon Jibiinkoka Gakkai Kaiho. 1996;99:314–319
- . Analysis of the causes of immediate unanticipated bleeding after pediatric adenotonsillectomy. Int. J. Pediatr. Otorhinolaryngol. 2003;67:341–344
- . Tonsillectomy and adenoidectomy. In: Hotaling AJ, Stankiewicz JA editor. Pediatric Otolaryngology for the General Otolaryngologist. New York, Tokyo: Igaku-Shoin Medical Publishers Inc.; 1996;
- . Complication of outpatient tonsillectomy and adenoidectomy: a review of 3,340 cases. Ear Nose Throat J. 1990;69:155–160
- . Unusual complications of tonsil and adenoid removal. J. Laryngol. Otol. 1969;83:1159–1174
- . Posttonsillectomy taste distortion: a significant complication. Laryngoscope. 2004;114:1206–1213
- . Post-transplant lymphoproliferative disease in tonsils of children with liver transplantation. Int. J. Pediatr Otorhinolaryngol. 2001;58:113–118
- M.J. Reilly, G. Milmoe, M. Pena, Three extraordinary complications of adenotonsillectomy. Int. J. Pediatr. Otorhinolaryngol., doi:10.1016/j.ijporl.2005.09.023, in press.
- . Fulminant post-tonsillectomy haemorrhage caused by aberrant course of the external carotid artery. J. Laryngol. Otol. 2005;119:655–657
- . An aberrant artery as a cause of massive bleeding following adenoidectomy. J. Laryngol. Otol. 2002;116:299–300
- . Lingual artery embolization for severe and uncontrollable postoperative tonsillar bleeding. Ear Nose Throat J. 2001;80:208–211
- . The vidian artery in childhood tonsillar hypertrophy. AJNR Am. J. Neuroradiol. 1988;9:141–143
- . Amigdalectomía y adenoidectomía. In: Paparella MM, Shumrick DA editor. Laringología. Madrid: Editorial Médica Panamericana; 1991;
- . Visual loss following tonsillectomy. Arch. Otolaryngol. 1968;87:436–438
- . Onset of paroxysmal myoglobinuria after tonsillectomy and adenoidectomy. Clev. Clin. Q. 1967;34:159–162
- . A severe late tonsillar hemorrhage 2 months postoperatively. Laryngol. Rhinol. Otol. (Stuttg). 1988;67:431–433
- . Extracranial internal carotid artery pseudoaneurysm. Int. J. Pediatr. Otorhinolaryngol. 2005;69:1135–1139
- . Arterial dissection complicating tonsillectomy. Dev. Med. Child Neurol. 2003;45:638–639
- . Tumors simulating neoplasia or of questionable neoplastic classification. In: Hyams VJ, Batsakis JG, Michaels L editor. Tumors of Upper Respiratory Tract and Ear. Washington, DC: Armed Forces Institute of Pathology; 1988;
PII: S1871-4048(06)00033-5
doi:10.1016/j.pedex.2006.01.009
© 2006 Elsevier Ireland Ltd. All rights reserved.
Volume 1, Issue 2 , Pages 119-122, June 2006
