the history of arachne through historic descriptions of … · story of arachne and of the main...

Historical Vignette

The History of Arachne Through Historic Descriptions of Meningiomas with Hyperostosis:

From Prehistory to the Present

Andrei Ionut Cucu1, Claudia Florida Costea2, Antonio Perciaccante6, Alexandru Carauleanu3, Serban Turliuc4,Bogdan Costachescu5, Ion Poeata5, Mihaela Dana Turliuc5

-BACKGROUND: Intracranial meningiomas are brain tumors that have prob-ably been known the longest, largely because of the occasional production ofgrotesque cranial deformities that have attracted the attention and interest ofhumankind. Because of the tendency of some intracranial meningiomas to causeskull deformation and thickening, these tumors have given rise to variousspeculations and theories related to their origin, starting in prehistoric times upto the present.

-METHODS: From the Steinheim skull and “pharaonic meningiomas” to thefirst meningioma monograph and the first explanations of Harvey Cushingregarding the mechanism of hyperostosis, this review aims to weave again thestory of Arachne. We identify the main contributors who have tried to understandand explain the tendency of some of these tumors to cause hyperostosis or otherskull bone involvements.

-CONCLUSIONS: The contribution of neurosurgeons or pathologists over thecenturies is of undeniable importance and is the basis for understanding futuremolecular mechanisms.

Key words- Hyperostosis- Meningioma- Skull deformities

From the 1Neurosurgery Clinic, “Prof. Dr. N. Oblu” EmergencyClinical Hospital, Iasi, Romania; Departments of2Ophthalmology, 3Obstetrics and Gynecology, 4Psychiatry,and 5Neurosurgery, “Grigore. T. Popa” University of Medicineand Pharmacy, Iasi, Romania; and 6Department of Medicine,“San Giovanni di Dio” Hospital, Gorizia, Italy

To whom correspondence should be addressed:Claudia Florida Costea, M.D., Ph.D.[E-mail: [email protected]]

Citation: World Neurosurg. (2019) 128:37-46.https://doi.org/10.1016/j.wneu.2019.04.199

Journal homepage: www.journals.elsevier.com/world-neurosurgery

Available online: www.sciencedirect.com

1878-8750/$ - see front matter ª 2019 Elsevier Inc. All

INTRODUCTION

Meningiomas are brain tumors that haveprobably been known the longest, largelybecause of the occasional production ofgrotesque cranial deformities1 that haveattracted the attention and interest ofhumankind. Because of the tendency ofsome intracranial meningiomas to causeskull thickening and deformation,throughout the history of humanity, thesetumors have left traces in the form ofhyperostosis that gave birth to the earlytheories related to their origin, startingwith the prehistoric period. From anarcheological perspective, we may say thatmeningiomas were primarily identified asa result of changes caused to theectocranial table,2 which were truepaleopathologic hallmarks of theselesions. From this point of view, 11intracranial meningiomas withhyperostosis with Paleolithic origins wererecorded worldwide (Table 1),3 and therarity of these tumors may be accountedfor by the fact that the mean lifeexpectancy for adults before the end ofthe medieval period was considered to be

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Table 1. Paleopathologic Examples of Meningiomatous Hyperostosis, According toOkonkwo and Laws3 and Anderson4

Location Date Gender Age (years)

Steinheim (Germany)5 365,000 BC F 25e30

Stetten (Germany)6 32,500 BC ? Adult

Chavina (Peru)7 Before contact F Middle age

Paucarcancha (Peru)8 Before contact M Elderly

Chicama (Peru)7 ? M Adult

St. Nicholas Island (California, USA)9 Before contact M Elderly

Helouan (Upper Egypt)10 3400 BC (I Dyn) M 40e60

Meydun (Upper Egypt)10 1500 BC (XX Dyn) M 50e70

Radley (Oxfordshire, United Kingdom)11 100 AD (Roman) F Adult

Chernovski (Alaska, USA)12 1000 AD M 40

Rochester (Canterbury, United Kingdom)4 1400 AD (medieval) F 35e50

F, female; M, male.

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ANDREI IONUT CUCU ET AL. HYPEROSTOSIS DESCRIPTIONS IN INTRACRANIAL MENINGIOMAS

anteroposterior size of 51 mm, amediolateral size of 43 mm, and a depthof 25 mm; its volume was 29 mL, thesize of a mean meningioma diagnosednowadays.5,6 In the Pleistocene era, thesetumor sizes, in conjunction with thesmall Steinheim cerebrum of only 1100e1200 mL, smaller than the modern brainof 1300e1800 mL, would have causedcontinuous headache, significanthemiparesis, and death.5

Ancient Egypt and PharaonicMeningiomasIn the ancient Egyptian medical papyridiscovered, there are no indications ofhead tumors with bone involvement.

Figure 1. (A) Steinheim city center at the time of theHomo steinheimensis skull (adapted, Urmenschmuse

38 www.SCIENCEDIRECT.com

However, tumors and tumorlike lesionswere not uncommon among the Pharaohs’people and they were mentioned espe-cially in the Ebers Papyrus, most probablypointing to alterations of the soft tissue.17

Discovered in 1862 between the legs of amummy in the necropolis of Theben, the20.23 m of the Ebers Papyrus containedmedical information, includinginformation about central nervous systemconditions.18,19

The earliest case of hyperostotic lesionof the skull from ancient Egypt wasdiscovered during the Royal Excavations atHelouan. The skull had a hyperostoticlesion in the right parietal area, whichinvolved both the inner and outer tables,

discovery of the Steinheim skull. (B) Replica ofumSteinheim) (public domain)

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showing the presence of a parasagittalmeningioma, and belonged to the FirstDynasty of Ancient Egypt (c. 3100e2890BC).10,20,21

A later case from the 20th Dynasty ofAncient Egypt (c. 1186e1069 BC) showed abeautiful specimen of calvarium, with anextensive honeycomb lesion of the rightparietal area extending to the left parietaland frontal areas.10 These AncientEgyptians, in their attempt to achieveimmortality for their Pharaohs andimportant officials, kept importantinformation about the diseases theyexperienced throughout their lives,22 alsoproviding information about “pharaonicmeningiomas."As concerns the treatment of head tu-

mors, Herodotus tells us that in ancientEgypt there were specialists of thehead23,24 and the remedies for these typesof tumors included excision with a knife,local paste applications, burning with red-hot irons, spells, or leaving the swellinguntreated.25,26 We have no documentary orphysical evidence that in ancient Egyptdoctors recognized or treated intracranialmeningiomas or other brain tumors.24

The oldest record of what could be ameningioma in the existing literature be-longs to Felix Platter (1536e1614), profes-sor at the University of Basel in Switzerland.In his book Platerus Observations in Hominis,published in 1614,27,28 Platter described thetumor as follows: “a round fleshy tumor,like an acorn. It was hard and full of holes,and as large as a medium-sized apple. Itwas covered by its own membrane andentwined with veins. However, it was freeof all connections of the matters of thebrain so much that when it was removed byhand, it left behind a remarkable cav-ity.”29,30 Despite the absence of hyperos-totic changes, the tumor described byPlatter is still a remarkably importantfinding in the history of meningiomas.Almost 100 years later (i.e., in 1730), also

on the old continent, the German JohannSalzmann (1672e1738) drew the earliestknown illustration of an intracranial me-ningioma with hyperostotic changes31

(Figure 2A). It was a left frontoparietaltumor with intracranial and extracranialcomponents. The case was called“Exostosisseu ex crescentia crania osseo-spongiosa,” and it was reported inCollegium Naturae Curiosum,31 the firstmedical and natural science journal, and in

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Figure 2. (A) Earliest known illustration of a meningioma,reported by Johann Salzmann in 1730, showing both intracranialand extracranial components of the tumor (adapted fromRefs.31,32 and the Biodiversity Heritage Library. Contributed by

the National History Museum Library, London, www.biodiversitylibrary.org). (B) The Kaufmann-Heister-Crellius casewith fungating meningioma (adapted from Refs.28,33). (C)Laurence Heister (1683e1758) (public domain).

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the present-dayGermanAcademy ofNaturalSciences Leopoldino.3,34

Professor of anatomy and pathologicanatomy at the University of Strasbourg,Johann Salzmann was interested inneuropathologic cases, of which hedescribed a few.35,36 One may notice inTabula VI (Figure 2A), which shows themeningioma, that both intracranial andextracranial components of this tumorare seen. As for the patient’s symptoms,they were insidious, lasted for >4 yearsand were dominated by the intracranialhypertension syndrome. This situationwas mainly determined by theintracranial component of the tumor,and Salzmann reconstituted meticulouslythe course of illness.35 As Salzmannreported in his article, in March 1727,after he had drunk some water, the 43-year-old man experienced severe abdom-inal pain, followed by a seizure, and sud-den death. Because of this sudden death,he was believed to have been poisonedand a forensic autopsy was performed.During autopsy on the left frontoparietal


side of the skull, a large tumor with extraand intracranial components was detec-ted.35 At the end of the report, Salzmannmentions that the tumor was soft,osseospongious, left an impression onthe underlying structures, and displacedthe dura and dural blood vessels withoutinfiltrating the brain and without duralinvolvement.34,35

The original report, written in Latin,provides a unique insight into the naturalhistory of an untreated calvarial ectopicmeningioma of that time.32 AlthoughSalzmann does not say anything abouttumor histology, in a report published in2004, Kompanje34 discusses the possibilitythat the tumor described by Salzmann mayhave been osteogenic sarcoma, metastaticcarcinoma, Ewing sarcoma, or giant celltumor. Considering the slow progressionof the tumor over at least 4 years, the largesize of the tumor with minor and insidioussymptoms extending over several years, theextracranial and intracranial componentsof the tumor, and the compressive effecton the underlying brain, the contemporary

GUST 2019 www.journals.

investigator is inclined to think it was aslow-growing benign process, most likely aprimary extradural meningioma. Moreover,Kompanje considers Salzmann’s case as oneof the first morbid descriptions of the find-ings during autopsy of a patient with me-ningioma ever reported in the scientificliterature, and the first reported case of arare primary extradural calvarial meningi-oma that provides information on the nat-ural history of the tumor.34 The next case ofcalvarial meningioma was reported muchlater, in 1932, by Bernard J. Alpers andReed Harrow from the University ofPennsylvania in Philadelphia.37

“De Tumore Capitis Fungoso” of aPrussian Soldier and a Spanish NoblemanSeveral years after the report of JohannSalzmann’s case in Collegium Naturae Curi-osum, also in Germany, in 1743, in Helm-stad, the surgeon and anatomist LaurenceHeister (1683e1758) (Figure 2C) markedthe beginning of the surgical treatmentof intracranial meningiomas.38 One ofDr. Heister’s patients was a 34-year-old

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Figure 3. (A) Antoine Louis (1723e1792). (B) “L’Ecole deChirurgie en construction” (1733). Académie Royale de

Chirurgie. Hubert Robert; Carnavalet Museum, Paris (publicdomain).

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Prussian soldier with an impressively largemeningioma, located at the vertex leveland having a significant extracranialcomponent (Figure 2B). Heister appliedcaustic lime to the tumor, but the patientdeveloped a sepsis infection andsubsequently died. The autopsy wasperformed by Johann Friedrich Crellius(1707e1747), who showed the tumor tobe “de tumore capitis fungoso.”38,39

Johann Friedrich Crellius was an anat-omy and physiology professor at Witten-berg and later in Helmstedt, payingspecial attention to pathologic anatomy.40

He wrote about the Prussian soldier’sexostotic meningioma in 1755, in hisdissertation thesis, together with JohannPhilipp Kaufmann, “De tumore capitisfungoso post cariem crania ex orto.” Thecase was to be later included in themonograph “Mémoire sur les tumeursfongueuses de la dure-mère,” publishedin 1774 by Antoine Louis.41

However, about a century before Heist-er’s surgical treatment, the Italian surgeonand anatomist Marcus Aurelius Severinus(1580e1656), author of Zootomia (the firsttreatise on comparative anatomy) per-formed the first successful surgical extir-pation of a brain tumor, so-called “fungustumor of the dura madre,” probably ameningioma.42 In his treatise “DeMedicina efficaci” (1646), Severinusdiscussed the operation he performed on


the skull of a nobleman of the Spanishcourt and of the house of the Avalos whohad intolerable headaches. Thenobleman consented to be subjected totrepanation of his skull, and Severinusfound a “fungous excrescence” under thebones. So, he proceeded to destroy theexcrescence, providing a definitive cureof the patient’s headaches.43

First Meningioma MonographWith the French Enlightenment, the or-ganization of medical sciences in WesternEurope started, as a result of intellectualproductivity improvement and ever higheracademic competition during the reigns ofLouis XV and Louis XVI.38,44 In the finalyears of the French Enlightenment, theFrench military surgeon Antoine Louis(1723e1792) (Figure 3A) produced the firstdocumented reference dealing exclusivelywith meningiomas.45

Antoine Louis was the descendant of afamily of surgeons andwas interested in thesurgery of the nervous system,38 especiallyof dural tumors, which he called“fongueuses de la dure-mère” (fungoidtumors of the dura mater),41 beingconvinced that intracranial meningiomasdevelop from the dura mater. However, itis not known whether Louis reached thisconclusion on the basis of his surgicalexperience or relying exclusively onautopsy studies.38,41


The name suggested by Louis, “fon-gueuses de la dure-mère,” was later to berecognized as the first important attempt toname meningiomas.3,38 He had his report,“Mémoire sur les tumeurs fongueuses de ladure-mère,” published in Mémoires de l’Aca-démie Royale de Chirurgie (Figure 3B) in 1774.38

In thismonograph, Louis reported 20 cases,most of which had been gathered in theliterature of the latter part of theseventeenth century and the eighteenthcentury up to the time of publication.88 Healso described and illustrated several casesof skull meningiomas eroding the boneand also having extracranial extension.41

Louis diagnosed these tumors basedprimarily on skull deformations, butobviously he also relied on neurologicsymptoms such as headache, weakness ofthe limbs, convulsions, or visual defects.46

Louis might have included under the termfungal tumors of the dura not onlymeningiomas but also metastases andsyphilitic osteomyelitis.47

The treatment of these tumors in Louis’time was the destruction of the tumor byapplying caustic solutions (e.g., potash),which was catastrophic in most cases,whereas other medicines, such as rosehoney, wine, or herbs, were ineffective.Nevertheless, Antoine Louis recom-mended the surgical removal of tumors,about which he also wrote an essay in1774.46

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The Most Beautiful Illustrations ofMeningiomasBeginning with the early nineteenth cen-tury, many doctors, among them JeanCruveilhier, have attempted to fill the gapthat appeared between a developingmodern science and an empirical andageing conception of medicine inheritedfrom antiquity and the Middle Ages.48

Being a pioneer in pathologic anatomyand general surgery, the Frenchman JeanCruveilhier (1791e1874) (Figure 4A) alsohad important contributions to researchon cancerous tumors of the meninges.47

Before him, his fellow countrymanAntoine Louis (1723e1792) provideddetailed descriptions of what he calledthe fungal tumors of the dura, whichincluded a variety of pathologic entitiessuch as syphilitic osteomyelitis andmetastases and not just meningiomas.47,50

Cruveilhier considered meningiomas tobe cancerous tumors of the meninges anddepending on their origin, he distinguished2 main types: tumors originating in theexternal layer of the dura, which tended togrow toward the cranial bone, which theyprogressively eroded, and tumors arisingfrom the internal layer of the dura, whichgrew inward and compressed the brain. Healso described a third rare type that he

Figure 4. (A) Frenchman Jean Cruveilhier (1791e1874invading the cribriform plate (adapted from Refs.47,49


believed originated in the arachnoid layer.47

As for bone involvement, he drewnumerous figures in his L’AnatomiePathologique du Corps Humain,49 perhaps themost beautiful medical book ever printed,illustrated with colored lithographs andmade from his extensive collection ofspecimens as professor of pathology(Figure 4B).3 The chapter on meningiomaswas called “Des Tumeurs Cancereuses desMeninges,” reflecting Cruveilhier’s beliefthat meningiomas were malignant tumors.3

Because of his encyclopedic descriptivework, Cruveilhier played a major role inthe birth of the anatomoclinical method,particularly in neurosurgery and neurologyduring the nineteenth century, towardevidence-based medicine.47

The First Successful Resection of aMeningiomaStarting with the early nineteenth century,in surgery of central nervous system tu-mors, the Italian school of surgery repre-sented by several Italian surgeons startedto advise that intracranial tumors shouldbe completely and promptly removed.51

Also, newly discovered knowledge inneuroanatomy and neuroscience startedto shape the discipline of neurosurgery

). (B) Large anterior skull base meningioma).


and neurology in the medical schoolsand hospitals of Europe.52,53

In this regard, the Italian Zanobi Pecchioli(1801e1866) was assigned the importantrole of having been the first to successfullyremove a meningioma.54 Pecchioli was aProfessor of Clinical Surgery and OperativeMedicine at Modena and Siena Universityand, in 1847, he reported his surgical seriesof 1524 surgeries performed over 16 years,from 1832 to 1847, 16 of which involvedskull trepanation.One of these surgeries was conducted in

Siena on July 29, 1835, and it involved theresection of a meningioma.38,54 Although itwas not reported in the literature byPecchioli, the case was described by theeditorial board of the Nuovo Giornale deiLetterati-Scienze in 1838.55 The tumor locationwas the right sinciput area. It was large, andthe entire tumor surface was ulcerated,which was proved by surgery to haveoriginated in the dura mater and to beeroding the skull.56 The 45-year-old patienthad long experienced severe headache thatwas exacerbated by digital compression ofthe mass.38,54 Pecchioli removed the masswith the dural insertion, “thus leaving theunderlying arachnoid exposed in two largezones.”56 The tumor was a large ulceratedcranial outgrowth and the extensivepostresection area took a long time to heal,with cambric soaked in sweet almond oil,and with the patient needing 4 months torecover.56 Five years later, in 1840, thedescription of the surgery was chosen forthe competition for the chair of surgery atthe University of Paris.55-57

Andrea Vaccà Berlinghieri (1772e1826)and Zanobi Pecchioli (1801e1866) sug-gested several surgical steps to remove“sarcoma” or “fungus” of the dura mater(meningioma), namely craniectomy ofbone flap with 5 or 6 burr holes, tumorand dura mater resection using a knife andtying of the cut vessels, and meningealartery occlusion at the edge of the bone, ifdamaged during surgery.51,56,57

One of the First Attempts at Skull BaseSurgeryOn November 26, 1841, the Venetian TitoVanzetti (1809e1888), professor of surgery atKharkov and surgeon for the Pope, exposedand removed a skull base tumor, which wasprobably a meningioma.58 The case wasdocumented 3 years later, in “Quelquesobservations pratiques recueillies à la


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Clinique Chirurgicale de l’UniversitéImpériale de Kharcoff (Russie)”59,60 andrepresented “a case of a bone cyst on theright side of the head that had destroyedthe base of the skull without affectingcerebral function.” The tumor proved to bea solid and massive tumor with intracranialgrowth, and during the operation, he hadto change his original surgical plan for abone cyst resection, because of theevidence of massive intracranial invasion.61

Although the patient survived theoperation, he died of an infection on the32nd day and his autopsy confirmed thehuge size of the injury and important skullbase invasion.56 Although Vanzetti did notreport the histology of the lesion, hisoperation can certainly be considered oneof the first attempts at skull base surgery,61

which probably involved a meningioma.58

“Omnis Cellula e Cellula”A few years later, in 1864, the renownedGerman doctor and pathologist RudolfLudwig Karl Virchow (1821e1920)(Figure 5A) described a psammoma of thedura (meningioma) located at skull baselevel, overlying an exostosis of thesphenoid bone, which was believed tohave developed as a consequence oftrauma.62 According to the figuresillustrating this case, the tumor extended

Figure 5. (A) Karl Virchow (1821e1920). (B) Illustrationskull base meningioma with exostosis of the sphenoid


behind the crista galli in the midline andthe bony exostosis apparently reached aconsiderable size37,62 (Figure 5B).Considered by many as the founder of

cellular pathology, Rudolf Virchow pro-vided the scientific basis for the modernpathologic study of cancer, also featuringin the history of medicine for his famous“Omnis cellulla e cellula” doctrine (i.e.,every cell from a cell).

Meningioma in the Hands of the ModernBrain Surgery PioneerSeveral years later, after Vanzetti’s success,in 1881, the Scottish surgeon WilliamMacewen (1848e1924) (Figure 5C),reported in The Lancet the successfulremoval of a left frontal meningioma.The case was of a 14-year-old girl with aswelling at the upper and inner part of theleft orbital cavity, with occasional frontalheadache and miotic left pupil with noresponse to light.63 Although the patienthad originally come to the doctor forcosmetic deformity, a few days afteradmission to the Glasgow RoyalInfirmary (Figure 5D), she experiencedseizures that progressed towardsecondary generalized seizures and life-threatening status epilepticus.64,65 Thegirl was sedated and under antisepticconditions, her skull was opened off the

of Virchow’s case ofbone (adapted from

Ref.62). (C) William Maceweduring Macewen’s time (in


right front lobe, and the detection of thetumor location was aided by hyperostosisof the skull above the left orbit.64-66

In the report in 1881 in The Lancet,Macewen described the surgical procedurein detail, but we focused only on thedescription of the bone infiltration by thetumor: “It was about the size and shape of akidney-bean [the tumor], its flat surfacelying against the roof of the orbit, andextending inwards under the orbital plateof the frontal. It seemed firmly fixed to theperiosteum, and it had a fibrous feeling.[.]. The bone underneath this littletumour was found to be rough and impar-ted a softer feeling to the finger than usual.[.] The skull was thicker than normal, andattached to the under surface of the discwas a tumour of a gummatous aspect.”29

After the surgery, Macewen noted that“after recovering from the influence of theanaesthetic, the convulsions did notreturn” and “her intelligence becameperfect.” Thus, on her discharge fromhospital, the patient was in goodcondition.29

At around the same time, in Turin,Italy, in 1883, the Italian surgeon FilippoGiacomo Novaro (1843e1934) operated ona parasagittal frontal hyperostosing me-ningioma.67 Just like in Vanzetti’s case,the diagnosis was set based on the bone

n (1848e1924). (D) Glasgow Royal Infirmarythe background of the image) (public domain).

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irregularities apparent from the outside,which were associated with epilepticseizures. In this case, Novaro resectedthe superior sagittal sinus that wasinfiltrated, but the patient died 1 hourafter the surgery56 as a result of acuteanemia caused by a profuseintraoperative hemorrhage from thediploe and superior sagittal sinus.54

Hyperostosis in DetaileFirst CaseIn 1899, Spiller reported in detail the firstcase of meningioma with hyperostosis thatenlarged the left side of the head. The casewas presented at themeeting of the Sectionon General Medicine of the College ofPhysicians of Philadelphia and was subse-quently reported together with Stern andKirkbride, the hospital’s pathologist. Theabstract of the report published in TheMedical Record68 claimed that: “the case wasthat of a man who, sixteen years previously,had fallen and injured the left side of hishead. After eight years a swelling wasnoticed in this situation, and several yearslater paresis in the right lower extremity,and later in the right upper extremity also.Headache and vomiting were superadded,speech became paraphasic, and aconvulsion occurred. On ophthalmoscopicexamination bilateral optic neuritis wasfound. Operation was decided upon; asegment of greatly thickened andinfiltrated bone was removed from thecalvarium, and a new growth found in thecortex of the left motor area. Hemorrhagewas profuse and death occurred a fewhours after the operation from loss ofblood and shock. On histologicexamination the growth was shown to bean endothelioma [meningioma].”Spiller drew attention on the contribu-

tion of head trauma to the occurrence ofhyperostosis, although the belief thatintracranial meningiomas were caused byhead injuries dates back to the eighteenthcentury, more precisely to Louis Antoine’stime. Spiller also reported 2 cases withprevious history of head trauma, arguingthat “it is not improbable that these in-juries were at least the predisposing causeof the bony proliferation.”69

During the following years, similarcases were reported by Brissaud and Lér-eboullet70 and later by Parhon andNadjede,71 who reported in 1905 the caseof a 63-year-old patient with bonyenlargement and an underlying tumor in


the right front lobe, the size of a smallorange.With the development of intracranial

radiology initiated by Bollici and Obici in1897,33,38 a few years later, in 1902, Pfahlerand Mills documented in Philadelphia thefirst radiologic description of a meningi-oma with some bone skull changes: “anexposure of four minutes [was] made witha moderately hard vacuum. A negative wasobtained which showed good detail of allthe structures. A large shadow lying be-tween the coronal suture and the posteriormeningeal artery corresponded to the areain which Dr. Mills had located thetumor.”38,72

Harvey Williams Cushing, the Father ofMeningiomasOn the American continent, in 1922,Harvey Cushing (1869e1939) first intro-duced the term meningioma to describe agroup of tumors that arose from themeninges of the brain and clarified thenumerous confusions and histopathologicnames used before to define this tumor.73

Cushing also studied hyperostosis relatedto intracranial meningiomas andreported a series of 20 cases ofmeningiomas with bony hyperostosis. Inevery case, Cushing noticed that tumorcells were found in the thickened boneand also pointed out that meningiomasen plaques were more frequentlyassociated with hyperostosis than weremeningioma en masse and bonyhyperostosis.73 The term meningioma“en plaque” was introduced by Cushingand his collaborator Eisenhardt todifferentiate these meningiomas from themore common form called meningioma“en masse.”33

Moreover, Cushing believed that thetumor cells were forced into the bonecanaliculi by increased intracranial pres-sure: “under the influence of intracranialpressure, the tumor cells. becomecrowded into and through the vasculardural spaces, and finally into the canaliculiof the bone. As a consequence, the bonebecomes rough, with subsequent osteo-blastic proliferation which causes hyper-ostosis. There can be little doubt that thethickening occurs in this way, but intra-cranial pressure may have nothing to dowith it, in view of the fact that the flatendotheliomas [meningiomas] which donot increase pressure are, as we have seen,


those which most often tend to invade thebone.”73

One of Cushing’s most famous patientswith intracranial meningioma was GeneralLeonard Wood (1860e1927), chief of Staffof the U.S. Army and a military surgeonhimself. Wood had sustained a head injuryfrom a low-lying chandelier in 1898,3 andseveral years later he noticed an exostosis,slowly increasing in size, occurring at thesite of the previous trauma at the vertex,right in the midline. In 1909, GeneralWood referred to Cushing, and after agenerally failed conservative treatment,Cushing performed 2-stage surgery onhim in 1910, at an interval of 4 days apart,and successfully removed the intracranialparasagittal meningioma. A few years later,in 1927, General Wood came again underCushing’s care because of his significantlyworsened condition. Although Cushingwas able to complete the extirpation of thetumor without any major complications,Wood died only a few hours after theoperation as a result of unexpected hem-orrhage that broke through into the ven-tricular system. Saddened, Cushing wouldconfess: “I’ve never lost a patient afteroperation that so upsetme. It was so near tosuccess. He was a great man.”74

Mechanism of HyperostosisLater after Cushing, Penfield and Phemistershowed the frequent bone involvement bymeningiomas,75,76 Phemister asserting that“the new bone is not tumorous in nature,and is merely the ossified stroma of theinvading endothelioma”.37,76 At around thesame time, in 1927, Grant was the first tosuccessfully remove the frontal hyperostosisof a bilateral parasagittal plaque tumor alongwith the sinus and the falx.77-79 At thebeginning of the twentieth century, someinvestigators such as Trossat, Martin,Dechauma, Weiser, Taylor, Kolodny, andWinkelman began to join their predecessorsin an attempt to explain the mechanism ofhyperostosis in intracranial meningiomas.37

Thus, the phenomenon of hyperostosisassociated with intracranial meningiomasstarted to be more thoroughly studied andalthough some investigators suggested thathyperostosis may be a manifestation oftumor invasion,73,80-87 others concludedthat these bony changes represent nothingelse but reactionary changes.88-92 Althoughsome investigators have recently arguedthat the existence of a neuropeptide


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serotonin influences bone metabolism93-97

or somatostatin receptor 2A expression,98

the research that has been carried out sofar has failed to identify the precisemechanism of hyperostosis andintralesional morphologic changes of thebone associated with meningioma.98 Thus,the exact molecular and biochemicalmechanism of hyperostosis in meningiomaremains to be explored.

CONCLUSIONS

Intracranial meningiomas have affectedhumankind since prehistoric times.Because of the ability of some intracranialmeningiomas to develop hyperostosis,which sometimes leads to skull thickeningand cranial deformities, they have attrac-ted attention, inspiring humans over cen-turies to explain their occurrence.Although the molecular mechanisms ofhyperostosis related to meningiomas arestill unknown, the contribution made byneurosurgeons and pathologists over thecenturies have been unarguably of majorimportance.

REFERENCES

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HISTORICAL VIGNETTE


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Conflict of interest statement: The authors declare that thearticle content was composed in the absence of anycommercial or financial relationships that could be construedas a potential conflict of interest.

Received 14 March 2019; accepted 22 April 2019


Citation: World Neurosurg. (2019) 128:37-46.https://doi.org/10.1016/j.wneu.2019.04.199

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The History of Arachne Through Historic Descriptions of Meningiomas with Hyperostosis: From Prehistory to the PresentIntroductionHomo steinheimensisAncient Egypt and Pharaonic Meningiomas“De Tumore Capitis Fungoso” of a Prussian Soldier and a Spanish NoblemanFirst Meningioma MonographThe Most Beautiful Illustrations of MeningiomasThe First Successful Resection of a MeningiomaOne of the First Attempts at Skull Base Surgery“Omnis Cellula e Cellula”Meningioma in the Hands of the Modern Brain Surgery PioneerHyperostosis in Detail–First CaseHarvey Williams Cushing, the Father of MeningiomasMechanism of Hyperostosis

ConclusionsReferences

the history of arachne through historic descriptions of … · story of arachne and of the main...

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