E x t r a m e d u l l a r y Hematopo ie s i s in a Subdura l H e m a t o m a

Case Report*

JOHN P. SLATER, M . D .

Department of Neurological Surgery, University of Vermont, Burlington, Vermont

I n f a n t s w i th a b n o r m a l l y large heads are of ten f ound to h av e s u b d u r a l h e m a t o m a s . T h e h e m a - t o m a fluid m a y v a r y f r o m old, acel lular fa in t ly x a n t h o e h r o m i c l iqu id to a f resh, b r igh t red, b loody fluid2 T h i s case r epo r t descr ibes an in- f a n t whose s u b d u r a l h e m a t o m a was a p p a r e n t l y a site of e ry thropoies i s .

C a s e R e p o r t

A 4-month-old baby girl was admitted to the Mary Fletcher Hospital on June 16, 1964, because her head

Received for publication January 7, 1966. * Presented at meeting of the New England Neuro-

surgical Society, February 19, 1965. Present address: Capt. John P. Slates, 05004778

106th General Hospital, A.P.O. 96503, San Francisco, Calif.

circumference had increased by 1�89 inches per month since April, 1964.

Examination. Her head circumference was 48.0 em.; chest circumference 38.0 cm. She was markedly pale. Laboratory studies revealed hemoglobin 4.4 grams per cent; hematoerit 19 grams per cent; 10,500 white blood per cu. ram., and a differential count of polymorphonu- clear leukocytes 48; lymphocytes 35; eosinophiles 7; monocytes 9; and basophiles 1. A "rare" nucleated red blood cell was seen; (less than 1 per 200 white blood cells). X-rays of the chest were normal. X-rays of the skull showed an enlarged head and obliteration of digital indentations.

Subdural taps were performed in the usual manner, through the lateral portions of the anterior fontanelle, on June 18, 1964. Thirty-five cc. of dark red bloody fluid were obtained from the right subdural space and 45 cc. from the left. A sample of this subdural fluid had a

T A B L E 1

Nucleated red blood cells in subdural fluids, June 18-July 8, 1964

Date

June 18

20 21 23 24 25 26 28 29 30

July 1 $

Total

Subdural Fluid

Fluid Aspirated from Left

44 CC.

40 cc.

35 ce.

40 ce.

80 cc.

29 ce.

15 cc.

15 CC.

10 cc.

259 cc.

Fluid Aspirated from Right

55 cc.

50 cc.

45 cc.

40 cc.

80 cc.

g0 cc.

25 cc.

15 cc.

270 cc.

Hematocrit

16%

2o%

17%

17%

17%

Nucleated RBC

2800/cu. mm. 71 NRBC

100 WBC

50 NRBC

1OO WBC

9O5/cu. mm. 55 NRBC

100 WBC 920/cu. mm.

62 NRBC

100 WBC

Peripheral Blood

Hematoeri t

19%

24%

29~o

29~o

26%

NRBC=Nu c lea t ed red blood ceils W B C = W h i t e blood cells.

~11

~1~ John P. Slater

Fro. 1. Photomicrographs a t same magnification, a. A single pronormoblast ; b. Two polychromic nornmblasts and one or thochromie nornmblas t .

to ta l cell count of 1,670,000/cu. nun; R B C 1,666,000/ cu. m m ; W B C 3,900/cu. m m ; 71 nucleated red blood cells per 100 white blood cells were counted. There were, therefore, approximate ly 2,800 nucleated red blood cells per cubic mil l imeter of subdural fluid. The differential white blood count showed 9 polymorpho- nuclear cells, 78 lymphocytes , 4 monocytes , 7 eosino- philes, 1 basophile, and 1 blast form.

The pat ient was t ransfused with 1~0 cc. of whole blood on June 18, 1964. Complete blood count per- formed after t ransfus ion showed hemoglobin 6.8 grams %; hematocr i t 24%; red blood cell count ~,9~0,000/ cubic ram; white blood cell coun t 10,900/cubic m m ; polymorphonuclear cells 41; lymphocytes 41; mono-

cytes 8; eosinophiles 10. The reticulocyte coun t was 11.1%. No nuclea ted red blood cells were identified on this or any subsequen t peripheral blood smear.

Table 1 t abu la tes the subdura l taps in the succeeding ~1 days . E igh t subdura l taps were performed on the right, r emoving a to ta l of s cc. Nine subdura l taps were performed on the left, removing an addi t ional 259 cc. The hematocr i t of the subdural fluid remained sta- t ionary a t 17% in spite of the repeated subdural aspira- tions. Nuc lea ted red blood cells remained in high concen- t ra t ion, 55 nuclea ted red blood cells per 100 white blood cells or 905 nucleated red blood cells/cubic ram.

N u m e r o u s very early red blood cell precursors were seen on smear of the pa t i en t ' s subdural fluid (Fig. 1). Basophilic normoblas ts , erythroblas ts and red blood cells in mi tos is were identified. These red cell precursors are normal ly found only in bone marrow. At no t ime were such i m m a t u r e nucleated red cells found in the pa t i en t ' s per ipheral blood.

The pa t i en t ' s fontanel le remained concave after the Ju ly 8 th aspi ra t ion; therefore no fur ther aspirat ions were performed.

On J u l y 15, 1964, an open right common carotid angiogram was performed. Our pre-angiographie diag- nosis was bilateral subdura l hema tomas wi th hema- topoiesis. We wished to rule out the possibility of an ar ter iovenous ma l fo rma t ion or other vascular anomaly as the source of this ex t ramedul lary erythropoiet ic act ivi ty. Th i s t h o u g h t had its origin in the 19~8 work of Cushing and Bailey 2 in which they speculated t h a t the blood in cyst ic t umors of vascular origin (i.e. heman- gioblas toma) was formed in the tumor itself. The angiogram, done wi th cross compression, demon- s t ra ted a mass ive subdura l hema toma covering each hemisphere. The re was no evidence of t umor or vascular anomaly . Di la ted lateral ventricles were suggested by a wide sweep of the anter ior cerebral arteries.

Operation. I m m e d i a t e l y following the angiogram the pa t ien t was t a k e n to the operat ing room where the sub- dural h e m a t o m a s were explored through bur r holes. The scalp, skull and dura were normal. The outer mem- branes of the h e m a t o m a s were thin and the inner mem- branes even th inner . T h e hema tomas were entirely liquid and measured 3 era. deep on the left, and ~ era. deep on the r ight . The re was no vascular anoma ly seen. T he h e m a t o m a s were washed from the subdura l space by repeated r insing with saline. Distilled water was used in the final rinse. Tab le ~ contrasts the i m m a t u r e red blood coun t in the subdura l fluid with t h a t of the peripheral blood a t the t ime of surgery.

Postoperative Course. Postoperat ively the pa t ien t did well. Her head circumference still measured 48 cm. and the anter ior fontanel le remained concave. She was discharged f rom the hospi ta l Ju ly ~3, 1964.

l~our subsequen t hospital izat ions have been neces- sary for the t r e a t m e n t of the subdural hema tomas . Samples of the subdura l fluid were obtained on each of these admissions . On her second admission she had subdura l aspirat ions, bo th of which showed a str iking preponderance of eosinophiles, 78% and 96% respec- tively. Tab le 8 cont ras t s the eosinophile count in the subdura l fluid wi th t h a t in the peripheral blood. In order to be cer ta in these cells were not macrophages laden with iron, an iron s ta in was performed which showed no iron present . The fluid was otherwise wi thin the l imits usual ly described as typical of subdural hematomas .