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Home , Craniopharyngioma

Spontaneous Rhinorrhea due to Case Report

ROBERT L. CAMPBELL, iV[.D., WOLFGANG ZEMAN, M.D., AND JOHN JOYNER, M.D. Department of Neurological Surgery, Indiana University Medical Center, Indianapolis, Indiana

Rhinorrhea is a rare complication of intrasellar hours; 17 hydroxycorticoids was 3~ rag. per ~4 hours, tumors. We have found only 7 reported eases. The with a total volume of 3,580 to 3260 ml. first was reported by Gutsche in 1895. 5 His pa- The patient was successfully treated with massive tient died with meningitis, as did the others de- doses of penicillin, sulfadiazine and chloromycetin. On January 4, 1961, an blectroeneephalogram revealed a scribed by Boyd, ~ Friedman} and Sore and grade one left frontal dysrhythmia. Ophthalmological Kramer> Berger t reported rhinorrhea secondary evaluation was normal. On January 7, 1961, a pneumo- to an inoperable craniopharyngioma, and Kay encephalogram was considered to be compatible with a et aU and Norsa n described this complication lesion extending through the dorsum sellae with in- with . There is no instance on volvement of the interpeduncular cisterns. This impres- record where, as in this case, a pituicytoma has sion was confirmed by angiography on January 9, 1961, produced rhinorrhea. which demonstrated stretching and straightening of C-1. Case Report Operation. The patient's condition continued to im- L.E.D., a 36-year-old man, was admitted to the In- prove and surgery was performed on January 35, 1961. diana University Medical Center on December 37, Through a right frontal craniotomy, a cystic mass oc- 1960. Earlier that day he had been seen by his family cupying the was found and subtotally re- physician because of a severe retro-orbital of moved. The histologic diagnosis was pituicytoma. The one day's duration, with progressive restlessness and space in the sella was packed with muscle. disorientation. Postoperative Course. The patient had diabetes in- The patient had been markedly obese since he was 17 sipidus during the 1st postoperative week and remained years old. He had noticed watery drainage from the disoriented. His state improved rapidly during the 3nd nostrils for a period of 6 to 8 nmnths prior to admission. week. On the 18th postoperative day he was discharged His father had had a craniotomy for removal of an from the hospital. He remained on Dilantin, thyroid and intrasellar mass, diagnosed as a pituitary tumor; no cortisone medication. histologic material was available for review. On February 9, 1961, the patient was started on a Examination. The patient was 5 feet, 10 inches tall, radiotherapeutic program during which he received and weighed 317 lbs. Physical examination revealed a 6,000 r to the tumor area over a 5-week period. He did blood pressure of 180/70, pulse of 9% respiratory rate of well until May, 1961, when CSF rhinorrhea recurred. 33, and a body temperature of 104.6 ~ F. He was dis- He was placed on antibiotics and the amount of corti- oriented and thrashed about purposelessly. The neck sone was increased. was not stiff. Scalp hair was full, but body hair was Second Operation. On June 13, 1961, the region of the scant, especially about the face, axillary and pubic sella turcica was re-explored and a sinus tract was iden- regions. Gynecomastia was present to a moderate degree. tified extending into the ethrnoidal cells. This area was The genitalia were small, the penis being only ~ cm. in packed with oxycel, Gelfoam and muscle. length. The testes were descended. The postoperative course was uneventful, with excep- The neurological examination was normal, with the tion of mild persistent and bitemporal exception of disorientation as to time and place. The . The patient was discharged July 13, deep reflexes were hyperactive. 1961. His case has been followed for the past 5 years. Laboratory Data. At the time of admission, the white During this time he has completed a course in electrical blood cell count was 49,900. On lumbar puncture the engineering and is now gainfully employed. The bitem- opening and closing pressures were in excess of 600 ram. poral hemianopsia has persisted. of spinal fluid. The cerebrospinal fluid was grossly purulent; 90% of the ~3,650 white ceils in the fluid were Discussion polymorphonuclear. Culture of the spinal fluid pro- There were no clinical signs suggestive of an duced a growth of gram positive diplococci. Skull x- rays demonstrated an enlarged, asymmetrical, eroded intrasellar mass lesion. Instead, the absence of sella turcica. visual disturbances and the presence of adiposo- During the 3nd week of hospitalization, other labora- genital features suggested a hypothalamic process. tory data were obtained. The clear liquid nasal dis- The clinical diagnosis was further masked by the charge revealed a sugar content of 53 rag.%. The fast- acute bacterial meningitis. ing blood sugar was 100 mg.%. PBI was reported 3.6 In view of the fact that the patient's father also and 5.3 gamma on 3 different occasions. Analysis of 17 had an intrasellar , it is interesting to ketosteroids ranged between 172 and 30.8 rag. per ~4 note that Kjellin et al. s cited ~7 similar cases and Received for publication November 39, 1965. added 7 of their own. They suggested heredity ~08 Rhinorrhea Due to Pituicytoma ~09

FIG. 1. Photomicrograph of pituicytoma. Note large cell bodies, oval, elongated nuclei, and acidophilie cytoplasm. There are areas of vascularity but no hemorrhage or necrosis. as a factor, but did not elaborate on its mode of References operation. 1. BERGER, I. Cerebrospinal rhinorrhea associated Primary of the neurohypophysis are with craniopharyngioma and meningitis. Report of rare, a fact which probably accounts for the con- a case. Arch. Otolaryng., Chicago, 1944, 39:184-185. fusion which has prevailed in respect to the no- 2. BOYD,W. A ease of tumour of the pituitary body. menclature. 9 Generically these neoplasms are glio- Lancet, 1910, 2:1129-1181. mas of the neurohypophysis, 12'13 to which Stern- 3. FRIEDMAN, A. P. Sur le symptome de la liquor- berg applied the term "choristoma"2 5 Globus 4 thee nasale. Encdphale, 1932, 27:129-133. called r apparently identical growths "infundib- 4. GLOBUS, J. H. Infundibuloma. A newly recog- nized tumor of neurohypophysial derivation with a uloma". Harland suggested that these neoplasms note on the saecus vasculosus. J. Neuropath. exp. represent "granular-cell-myoblastoma". 6 The lat- Neurol., 1942, 1:59-80. ter term is not only confusing, but also incorrect, 5. GUTSCHE, P. Zur Pathogenese der Hypophy- as Liss and Kahn I~ produced reasonably good evi- sentumoren umd tiber nasalen Abfluss sowie das dence of the neurogenic nature of the tumor cells Verhalten des Liquor cerebrospinalis bei einer by tissue culture. Harland's terminology seems to Struma pituitomata. Int. Zbl. Laryng., 1895, imply a mesodermal origin for these tumors, s On 11:460. the strength of his observations, Liss proposed the 6. HARLAND, W. A. Granular-cell myoblastoma of the hypophyseal stalk. Cancer, 1953, 6:1184-1138. term "pituicytoma", which was reluctantly ac- 7. Kxv, S., LEES, J. K., and STOtrT, A.P. Pituitary cepted by Russell and Rubinstein. 1~ We favor ehromophobe tumors of the nasal cavity. Cancer, this terminology because of the extremely slow 1950, 3:695-704. progression of the neoplastic growth, a feature im- 8. KJELLm, K., MULLER, R., and ASTROM, K. E. plicit in the term pituicytoma, but not necessarily The occurrence of brain tumors in several members inferred by the designation . of a family. J. Neuropath. exp. Neurol., 1960. 19:528-587. Summary 9. LIMA, A., ANTUNES, L., and TOME, F. Pituicy- We have reported a case of spontaneous rhinor- toma or granular-cell myoblastoma of the pituitary rhea associated with a pituicytoma. The patient gland? J. Neurosurg., 1960, 17:778-782. 10. Lms, L., and KAHN, E.A. Pituicytoma. Tumor of had the signs and symptoms of an adiposo-genital the sella tureica. A clinieopathological study. J. syndrome of many years duration. The sella tur- Neurosurg., 1958, 15:481-488. cica was enlarged. There was no visual field defect 11. NoasA, L. Cerebrospinal rhinorrhea with pitui- prior to the operation. A pituicytoma was par- tary tumors. Neurology, 1958, 3:864-868. tially removed. 12. RUSSELL,D. S., and RUmNSTEIN, L.J. Pathology