According to Emge [3], in 1882 von Recklinghausen suggested to coin this type of pathology “adenomyoma uteri”, but gives no reference. What we know is that at the end of the nineteenth century, in 1895 and 1896 to be exact, the condition was clearly described by von Recklinghausen [4, 5] and Cullen [6], followed by Pick [7] and Rolly [8] in 1897. In a seminal book on adenomyoma published in 1908, Cullen [9] states that approximately 100 cases were described prior to his first paper of 1896.

In fact, the first description of a condition today recognised to be a form of adenomyosis is contained in an article published in 1860 by Rokitansky [10] in which he reported on three cases of “fibrous polyps of the uterus” and stated: “among them there are some in which glandular tubes are found”. In detailing his findings Rokitansky mentions that “In some rare cases the extension of the uterine glands occurred in both directions, i.e. both into the uterus cavity, as well as into the uterus parenchyma, such that the sloped bulge represents a plug of longitudinal fibrous appearance driven into, as it were, the uterine mass”.

Rokitansky’s description apparently misled later readers, even those almost his contemporaries, on two grounds: First, a polypoid structure protruding inside the uterus would hardly be considered an “adenomyoma”. Second, the name attributed to this lesion by Rokitansky was “cistosarcoma adenoids uterinum” and later “sarcoma adenoids uterinum polyposum”. Both names seemed to distract attention from his description as representing what we today call adenomyosis.

At any rate, the link between Rokitansky’s description and the discovery of adenomyosis is reiterated in a few recent articles describing “adenomyomatous polyps”. A literature search [11] identified 13 reports (mostly from Japan) of adenomyomatous polyps; structures that Raghavendra Babu et al. [12] defined as follows: “in addition to the usual features of endometrial polyps, they also contain a smooth muscle component”. Histologically they are composed of “endometrial glands intimately mixed with smooth muscle and thick walled blood vessels”. An interesting issue surrounds adenomyomatous polyps, because a Medline search for “polypoid adenomyomas” yields more than 100 articles (mostly of atypical forms); yet the descriptions lead to the clear conclusion that the same pathology is reported under two different titles.

At any rate, Rokitansky specifies: “The thick-walled uterus of an aged female showed this inter alia. On the left hand side under the mouth of the tuba was a swollen, about 1–2 long smoothly coated polyp of 11/2 diameter in the pedunculus, from 4–5 on the free end. A similar bisecting perpendicular section continuing into the uterine mass shows that the pedunculus penetrates to a depth of 4 into the uterine mass and stores a wedge driven right in to the uterine tissue; on its section along its length it has a fibrous appearance and can be torn into fibres in this direction; the arrangement of the fibres is determined by numerous extremely long glandular tubes held together by means of a core-rich connective tissue” [10].

A comprehensive definition of an adenomyoma was provided by Cuthbert Lockyer in 1918 [13]: “the term ‘adenomyoma’ implies a new formation composed of gland-elements, hyperplastic cellular connective tissue, and smooth muscle”. He added: “so far as the adenomatous elements are concerned, the same type of tumour-formation can be found also in the digestive tract (bowel and stomach), and some observers claim that analogous conditions can exist in the gall-bladder, in the kidney, and elsewhere”.

In spite of the clarity of Cullen’s early description [6], which he expanded in his 1908 book [9], the medical community investigating ‘mucosal invasions’ of abdominal organs, in general failed to identify them as being due to the heterotopic presence of uterine mucosa and the nature of glands found in adenomyomata remained controversial for a long time [see also Chap. 6]: in 1904 Schickele [14] attacked Cullen’s theory and argued that the mucosal growth was of mesonephric origin. He wrote “when I try to take an impartial view of published cases, I am compelled to state that the mucosal theory is not proved”. Indeed, the presence of multiple communications with the lumen of the tube, or with the endometrial cavity constituted no proof to his mind.

During the early part of the twentieth century, the controversy over the origin of the epithelial cells lining the ‘cysts’ found in adenomyomata continued. Ignoring Rokitansky’s conclusions, pathologists of the fame of von Recklinghausen [5] argued that adenomyomata were the result of displacement of Wolffian or mesonephric vestiges. He illustrated his theory of the origin of adenomyomata showing that glands were scattered along Wolffian remnants. The majority of pathologists and gynecologists then rejected the hypothesis that the glands they observed were “endometrial.”

As late as 1918, Lockyer [13] in the above-mentioned book “Fibroids and allied tumours”, in detailing the various theories on the origin of epithelial glands and stroma found in the pelvis outside the uterine cavity, was unable to resolve the question of their origin. He wrote: “nothing but the topography of the tumour, nothing but laborious research entailing the cutting of serial sections in great numbers, can settle the question as to the starting point of the glandular inclusions for many of the cases of adenomyoma”. Lockyer reviewed a series of cases which he considered of ‘serosal’ origin. Five cases were labelled ‘adenomyoma of the ovarian ligament’ and had been published previously [14–18] (with today’s knowledge these would be probably considered cases of deep endometriosis); he accepted Frankl’s theory [18] that these tumours arose from parts of the Wolffian system (medullary cord or duct). Lockyer [13] also reported that before the name was coined there had been clear descriptions of adenomyomata, the first being those made by Babes [19], who, in 1882, published a case of an intramural myoma containing cysts lined with “low cubical epithelium derived from embryonic germs” and by Diesterweg [20] who, the following year, described “two polypi of the posterior uterine wall containing cysts lined with ciliated epithelium and filled with blood”. At the time it was widely believed that epithelial cells found in adenomyomata (within and outside the uterus) had a Müllerian origin. In his 1908 book Cullen [9] mentions that, by 1884, some 100 cases had been reported by Schröder, Herr and Grosskopf, but provides no reference. Then, in 1893, von Recklinghausen published his first observations on adenomyomata (initially named by him “adenocysten” of the uterus) [21], followed by his 1895 publication [4] and his acclaimed book of 1896 [5] and divided adenomyomata into two classes: