CORRESPONDENCE to Ms Ryoko Sakai, Mr Osamu Kanda, Ms Kotomi Shibata, Ms Chie Nakashima, and Ms Chikako Matsuo for their technical assistance.
antibodies. However, so-called L a n g e r h a n s g r a n u l e s were n o t d e t e c t e d by e l e c t r o n microscopy. I n a r e c e n t l y r e p o r t e d case o f EC, histiocytes in t h e lesion also s h o w e d positive s t a i n i n g with anti-S-100 antibody. 17 T h e s e f i n d i n g s i n d i c a t e t h a t t h e histiocytes in EC a p p e a r to have characteristics i n t e r m e d i a t e b e t w e e n those o f L a n g e r h a n s cell histiocytosis cells a n d ordinary histiocytes; if so, in a b r o a d e r sense, EC may c o n s t i t u t e p a r t of t h e s p e c t r u m o f histiocytosis. H y p e r l i p e m i a is n o t always associated with EC. 1 A l t h o u g h the b o n e lesion in o u r p a t i e n t c o n t a i n e d a large a m o u n t o f c h o l e s t e r o l ester, l a b o r a t o r y study o f t h e s e r u m lipid s h o w e d n o a b n o r m a l findings, e x c e p t for slight elevation o f triglyceride. T h e s e f i n d i n g s suggest t h a t t h e disease s h o u l d n o t b e a t t r i b u t e d to a b n o r m a l systemic lipid m e t a b o l i s m , b u t t h a t it is c a u s e d by histiocyte p r o l i f e r a t i o n , with activated phagocytosis. Jaffe n states t h a t t h e a p p e a r a n c e o f foamy m a c r o p h a g e s seems to b e a n essential p h e n o m e n o n in EC b e c a u s e f o a m y m a c r o p h a g e s were t h e p r e d o m i n a n t f i n d i n g , a n d i n f l a m m a t o r y rea c t i o n was n o t as p r o m i n e n t in all cases r e p o r t e d . As stated previously, t h e r e was a n obvious d i f f e r e n c e in t h e a m o u n t of t h e lipid c o m p o n e n t in t h e tissues o f o u r pat i e n t a n d in t h e tissues o f t h e p a t i e n t r e p o r t e d by F u r u s e et al. 9 Because t h e histological a p p e a r a n c e a n d t h e radiological features were a l m o s t identical in these two patients, it is difficult to explain t h e d i f f e r e n c e s in t h e a m o u n t o f lipid. O n e possible e x p l a n a t i o n is t h a t activated histiocytes may have diff e r e n t p r e d i l e c t i o n s for t h e i n g e s t i o n o f lipid d e p e n d i n g o n s o m e u n k n o w n circumstances. A n o t h e r possibility is t h a t several d i f f e r e n t diseases c o u l d show similar s y m p t o m s a n d b e c o n s i d e r e d as t h e same entity. However, b i o c h e m i c a l analysis of t h e lesion has b e e n p e r f o r m e d in only two patients, including ours. T h e r e f o r e , f r o m such little i n f o r m a t i o n , it is difficult to draw a c o n c l u s i o n o n t h e type o f lipid p r e d o m i n a n t l y prese n t in m a c r o p h a g e s . F u r t h e r cytological e x a m i n a t i o n is req u i r e d to analyze t h e biological f u n c t i o n o f t h e histiocytes in this lesion i n c l u d i n g t h e activity o f phagocytosis.
REFERENCES 1. Lantz B, Lange TA, HeinerJ, et al: Erdheim-Chester disease. A report of three cases.J Bone Joint Surg 71-A:456-464, 1989 2. Miller RL, Sheeler LR, Bauer TW, et al: Erdheim-Chester disease. Case report and review of the literature. AmJ Med 80:1230-1236, 1986 3. Resnick D, Greenway G, Genant H, et al: Erdbeim-Chester disease. Radiology 142:289-295, 1982 4. Brower AC, Worsham GF, and Dudley Aid: Erdheim-Chester disease: Distinctivelipoidosis or part of the spectrum of histiocytosis?Radiology 151:3538, 1984 5. Hsu S-M, Raine L, Fanger H: Use of avidin-biotin peroxidase complex (ABC) in immunoperoxidase techniques: A comparison between ABC and unlabeled antibody (PAP) procedures.J Histochem Cytochem 29:577-580, 1981 6. Kato K, Kimura S, Haimoto H, et al: S-lO0,~(a/a) protein: Distribution in muscle tissues of various animals and purification from human pectoral muscle. J Neurochem 46:1555-1560, 1986 7. Folch J, Lee M, Sloane-Stanley GH: A simple method for the isolation and purification of total lipids from animal tissues.J Biol Chem 226:49%509, 1957 8. Lowry OH, Rosebrough NJ, Farr AL, et al: Protein measurement with the Folin phenol reagent. J Biol Chem 193:265-275, 1951 9. Furuse K, Minamizaki T, Yoshida H, et al: A case of Erdheim-Chester disease with hyperlipoproteinemia. Byouri to Rinshou (Japanese with English abstract) 9:1357-1363, 1991 10. Chester W: Uber lipoidgranulomatose. Virchows Arch Pathol Anat 279:561-602, 1930 11. Jaffe HL: Metabolic, degenerative and inflammatory disorders of bone and joints. Philadelphia, Lea and Febiger, 1972, pp 535-560 12. Simpson FG, Robinson pJ, Hardy GJ, et al: Case reports. ErdheimChester disease associated with retroperitoneal xanthogranuloma. Br J Radiol 52:232-235, 1979 13. Poehling GG, Adair DM, Haupt HA: Erdheim-Chester disease. A case report. Clin Orthop 185:241-244, 1984 14. Rozenberg I, Wechsler J, Koenig F, et al: Erdheim-Chester disease as malignant exophthahnos. BrJ Radiol 59:173-177, 1986 15. Tien RD, Brasch RC, Jackson DE, et al: Cerebral Erdheim-Chester disease: Persistent enhancement with Gd-DTPA on MR images. Radiology 172:791-792, 1989 16. Tien R, KucharczykJ, Newton TH, et al: MR of diabetes insipidus in a patient with Erdheim-Chester disease: Case report. AmJ Neuroradiol 11:12671270, 1990 17. Athanasou NA, and Barbatis C: Erdheim-Chester disease with epiphyseal and systemic disease. J Clin Pathol 46:481-482, 1993 18. EbleJN, RosenbergAE, Young RH: Retroperitoneal xanthogranuloma in a patient with Erdheim-Chester disease. AmJ Surg Pathol 18:843-848, 1994 19. MosleyJE: Patterns of bone change in the reticuloendotbelioses. J Mount Sinai Hospital (New York) 29:282-321, 1962 20. Molnar CP, Gottschalk R, Gallagher B: Lipid granulomatosis: ErdheimChester disease. Clin Nucl Med 13:736-741, 1988
Acknowledgment. We wish to thank Dr Shin-Ichiro Ushigome, Department of Pathology, The Jikei University School of Medicine, Tokyo; and Dr Takayttki Nojima, Department of Pathology, Kanazawa Medical University, Kanazawa, for helpful suggestions. We are grateful
CORRESPONDENCE T h e y m a d e a valid s t a t e m e n t a b o u t m a l i g n a n t m e l a n o m a a n d t h e i r p a t h o l o g i c a l pictures. I l e a r n e d m u c h f r o m this r e p o r t . I w a n t raise a q u e s t i o n a b o u t t h e x-ray film o f this h i p joint. As t h e a u t h o r s stated in t h e case r e p o r t , " A r o e n t g e n o g r a m o f t h e left h i p (Fig 1 left) revealed a p e r m e a t i v e a n d destructive radiolucent lesion involving t h e f e m o r a l h e a d . " T o me, t h e lesion is r a d i o p a q u e , for t h e following reasons: t h e f e m o r a l h e a d a n d n e c k is obviously a b n o r m a l in a r c h i t e c t u r e , its color is u n e v e n , a n d it is a m i x e d s h a d o w o f white a n d black. B u t obviously it has too m u c h white in its c e n t e r p o r t i o n , a n d it is n o t h o m o g e n o u s with s o m e small d a r k " h o l e s " o r " s p o t s " in t h e c e n t e r o f this white diseased s h a d o w w h i c h occupies t h e c e n t e r p o r t i o n o f t h e f e m o r a l h e a d a n d e x t e n d s i n t o t h e c e n t e r p a r t of t h e i n n e r f e m o r a l neck. So it is r a t h e r large in volume, a p p r o x i m a t e l y 2.5 c m in d i a m e t e r . Its i n n e r 1/3 p o r t i o n is o v e r l a p p e d with t h e acet a b u l u m r i m a n d its o u t e r 2/3 is outside t h e a c e t a b u l u m a n d is seen in t h e i n n e r n e c k p o r t i o n . B u t t h e o t h e r o u t e r a n d u p p e r small p a r t o f t h e f e m o r a l h e a d is clearly r a d i o l u c e n t a n d it h a s p r o b a b l y e s c a p e d t h e t u m o r invasion. T h e r e f o r e ,
Guidelines for Letters Letters to t h e E d i t o r will b e p u b l i s h e d at t h e disc r e t i o n o f t h e e d i t o r as space p e r m i t s a n d are subject to e d i t i n g a n d a b r i d g e m e n t . T h e y s h o u l d b e typewritten, double-spaced, a n d s u b m i t t e d in triplicate. T h e y s h o u l d b e limited to 500 words o r less a n d to n o m o r e t h a n five p e r t i n e n t references.
Read Film with Caution To the Editor:--I have b e e n a r e a d e r o f your j o u r n a l for m a n y years. I have g a i n e d m u c h k n o w l e d g e f r o m it. Recently, I r e a d a p a p e r by Dr. Philip J. Daroca, Jr, et al I with g r e a t interest. T h e y p r e s e n t e d a very i n t e r e s t i n g case with g o o d film illustrations, b o t h in p a t h o l o g y as well as in r o e n t g e n o l o g y .
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HUMAN PATHOLOGY
Volume 27, No. 1 (January 1996)
this lesion could not be interpreted as "radiolucent," but it is rather "radiopaque" in nature, and it is usually interpreted as osteoblastic. One more reason is that this osteoblastic lesion is congruent with the two dense spot lesions in the left ischium which were mentioned by Dr. Daroca and probably treated as another two small metastatic changes. Therefore they all represent the same pathological processes in this particular patient. I know the white and black shadows in film reading, but I d o n ' t know if the above opinion is correct or not.
were from girls and presented at birth. They were all histologically typical and showed characteristic large granular cells and no hyperplasia of the overlying epithelium. None showed immunoreactivityfor S100. Two showed immunoreactivity for CD68. One was negative for CD68, although macrophages within the tumor formed an internal positive control (see Fig 1). This tumor was the largest of the tumors measuring 2.5 cm in diameter. There are several recent reviews of granular cell tumors and congenital granular cell epulis, s'4 The histogenesis of congenital granular cell epulis is unknown. An origin from an undifferentiated mesenchymal cell is favored. 4 Their characteristic site on the lateral alveolar ridge, female preponderance, and S100 negativity separate them from granular cell tumors in other sites. The ultrastructural appearance of the granules in these tumors is similar to those in conventional granular cell tumors. The lack of CD68 positive staining in one case appears to be genuine, and not due to a technical or diagnostic problem. It indicates that as well as the different clinical features and negative staining for S100, variable CD68 immunoreactivity is a further difference between congenital granular eell epulis and conventional granular cell tumors.
LIAN BI HU, MD Visiting Chief in Diagnostic Roentgenology Staff Hospital Petroleum Control Department of Sichuan Province Zhong Xing Chang Chengdu, China 1. Daroca PJ, Jr, Reed RJ, Martin PC: Metastatic amelanotic melanoma simulating giant-cell tumor of bone. HUM PATHOL 21:978-980, 1990
Granular Cell Epulis To the Editor:--I was interested to see the article entitled, "Immunohistochemical Demonstration of the Lysosome-Associated Glycoprotein CD68 (KP-1) in Granular Cell Tumors and Schwannomas," by Kurtin and Bonin, 1 as there is debate about the histogenetic relationship between these tumors and congenital granular cell epulis. Review of the recent literature revealed one reference in which CD68 positivity was described in a congenital epulis. 2 I examined three congenital granular cell epulides from the files of the Bristol Royal Hospital for Sick Children. All
ADMAN CHARLES, MRCPath Department of Pediatric Pathology St. Michael's Hospital Bristol, UK 1. Kurtin PJ, Bonin DM: Immunohistochemical demonstration of the lysosome-associated glycoprotein CD68 (KP1) in granular cell tumors and schwannomas. HUM PATHOL 25:1172-1178, 1994 2. Tsang WYW, Chart JKC: KP1 (CD68) Staining of granular cell neoplasms: Is KP1 a marker for lysosomes rather than the histiocytic lineage? Histopathology 21:84-86, 1992 3. Kershisoik M, Batsakis JG, Mackay B: Granular cell Tumors. Ann Otol Rhinol Laryngol 103:416-419, 1994 4. Zuker RM, Buenechea R: Congenital epulis: Review of the literature and Case Report. J Oral Maxillot:ac Surg 51:1040-1043, 1993
Diagnostic Criteria of Molar Conditions To the Editor:--The criteria by which Cheville and associates t distinguish the various molar entities are skewed; they define hydropic degeneration as lacking cistern formation, although such change has been well documented in the hydropic abortus as best illustrated in a recent study,2 and again mentioned in the next article of the same issue) No wonder that the diagnostic accuracy remains poor 4 when pathological classification is imperfect. JACQUES F. LEGIER, MD Department of Pathology Riverside Regional Medical Center Newport News, VA 1. Cheville JC, Greiner T, Robinson RA, et al: Ploidy analysis by flow cytometry and fluorescence in situ hybridization in hydropic placentas and gestational trophoblastic disease. HUM PATHOL 26:753-757, 1995 2. Fukunaga M, Ushigome S, Fukunaga M, et al: Application of flow cytometry in diagnosis of hydatiform moles. Mod Pathol 6:35~359, 1993 3. Fukunaga M, Ushigome S, Endo Y: Incidence of bydatdiiform mole in a Tokyo Hospital. A 5 year (1989 to 1993) prospective, morphological, and flow cytometric study. HuM PATHOL 26:758-764, 1995 4. Howat AJ, Beck S, Fox H, et al: Can histopathologist, reliably diagnose molar pregnancy? J Clin Pathol 46:5994302, 1993
The above Letter was referred to the authors of the article in question, who offer the following reply:
FIGURE 1. Congenital granular cell epulis. Immunohistochemical staining with CD 68 antibody, showing negative staining of the granular cells, with positive staining of a macrophage.
To theEditor:--We appreciate Dr. Legier's comments. We are aware that hydropic degeneration in early pregnancy loss
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