Surgical Globetrotting

Chronic Granulomatous Mastitis: Diagnostic and Therapeutic Considerations

Aziz Firzah Azlina, M.S.,1 Zakaria Ariza, M.B.B.S.,1 Talib Arni, M. Path.,2 Abdullah Noor Hisham, M.S.1

1Department of Breast and Endocrine Surgery, Putrajaya Hospital, Precinct 7, 62250 Putrajaya, Malaysia2Department of Pathology, Kuala Lumpur Hospital, Jalan Pahang, 50586 Kuala Lumpur, Malaysia

Abstract. To review the clinical presentation, histopathological features,and optimal treatment of chronic granulomatous mastitis, the authors con-ducted a retrospective study of 25 women admitted to a teaching hospital inMalaysia between January 1998 and December 2000 who met the requiredhistologic criteria. The primary outcome measures were morbidity and re-currence of the disease. Thirteen patients presented with a breast massclinically mimicking breast cancer, and 12 patients had breast indurationand abscess formation. In addition, 8 of these patients had recurrent breastdisease. Clinical and imaging diagnosis has often been difficult and incon-clusive, so histopathology remains the optimal diagnostic tool. Of interest,50% of patients experience recurrences, and long-term follow-up is there-fore necessary. The authors concluded that, because chronic granuloma-tous mastitis is a rare benign breast condition that may be misdiagnosed asbreast carcinoma, complete resection should be accomplished wheneverpossible. Steroid therapy may be an adjuvant for optimal treatment.Awareness among surgeons and pathologists should also be emphasized toavoid unnecessary misdiagnosis and treatment.

Chronic granulomatous mastitis (CGM) is a rare benign breastcondition first described by Kessler and Wolloch in 1972 [1]. Theyreported five women with breast masses characterized by floridgranulomatous mastitis, which was not associated with trauma, spe-cific infections, or exogenous materials. To date, there are only ap-proximately 120 reported cases in the international literature [2].Although it is a benign entity, CGM clinically mimics breast cancerin terms of physical and radiological findings, often leading to di-sastrous consequences. At present, the definitive diagnosis can onlybe established and confirmed by histopathology. The aim of thisstudy was to review the clinical presentation of CGM and its histo-pathological features, and to outline the diagnostic and therapeuticconsiderations in the management of this potentially recurrentnonmalignant breast disease.

Patients and Methods

A retrospective study included 25 women who met the requiredhistological criteria of CGM and who were treated between Janu-ary 1998 and December 2000. This study was performed in a teach-

ing hospital; the patients were either referred from the OutpatientDepartment of the hospital or from peripheral government healthclinics with outpatient services. The diagnosis was confirmed eitherby core needle biopsy or fine-needle aspiration cytology for the sus-picious breast lesions and also from biopsy specimens taken fromthe abscess wall during drainage. Clinical data of the presentation,histopathology, and management were analyzed by review of medi-cal records. Patients who had history of cosmetic treatment by in-jection of silicone, liquid paraffin, or beeswax into the breast pa-renchyma (olegranulomatous mastitis) were excluded from thisstudy.

Follow-up information was obtained from clinical reviews atmonthly intervals (range, 2 to 11 months). The types of symptoms,severity, and duration were documented. The data collected werethen studied and the various parameters were compared retrospec-tively.

Results

Complete follow-up information for all patients diagnosed withCGM with regard to the clinical presentation, histology report, andmanagement was obtained for all 25 patients. The patients rangedin age from 21 to 47 years, with the exception of one patient whowas 74 years old. The median age was 36.5 years. There were 19Malay and 6 Indian ethnic patients in our series. The parity of thesepatients ranged from nil to six, with a mean parity of three. At thetime of presentation, one patient was 28 weeks pregnant and fivepatients were lactating. None of these patients were on any medi-cation or hormonal treatment.

All patients except one presented with a history of a painful pal-pable lesion in the affected breast. These lesions were unilateraland occurred with no tendency to any particular quadrant of therespective breast. The size of the mass ranged from 1 to 10 cm witha mean of 5.5 cm. There was also no tendency for any side of thebreast to be more involved (left 64%, right 36%). The mean dura-tion of symptoms was 2.0 months. At the time of presentation, 12women (52.0%) had a clinical impression of breast abscess. Theother 13 women in this study (48.0%) had a clinical impression of amalignant breast lump. More than half the women in our series hadalready been treated with a course of antibiotics with or without

Correspondence to: Abdullah Noor Hisham, M.S., e-mail: anhisham@pd.jaring.my

WORLDJournal of

SURGERY© 2003 by the Societe

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World J. Surg. 27, 515–518, 2003DOI: 10.1007/s00268-003-6806-1

drainage before presenting at our clinic. Of these, 11 (41%) hadreceived antibiotic therapy, and 4 (24%) had additional incisionand drainage procedure performed. However, despite treatment,these abscesses failed to resolve.

Of the 13 patients with the preliminary clinical impression of ma-lignant breast lesion, the preoperative diagnosis was made either byfine-needle aspiration cytology (FNAC) (n = 3) or core biopsy (n =9). Only one (33%) of the 3 patients who had FNAC achieved thediagnosis of CGM using the FNAC sample. The other two womenwho initially had FNAC done had a second biopsy (core biopsy),which later confirmed the diagnosis of CGM. These 2 patientseventually had wide excisions performed. The histology reportfrom the excision biopsy or incision of the abscess wall duringdrainage confirmed the diagnosis in all cases. Microscopically,there were areas of necrosis and acute and chronic inflammatorycells with foamy macrophages, epithelioid cells, and multinucleat-ed giant cells forming granulomas (Fig. 1).

There were no caseations; however microabscesses were some-times present. There were no malignant cells, fungi, or acid-fastbacilli seen. All patients had pus cultures, none of which showedany growth. In most cases, however, the cultures were taken afterthe patients had been started on antibiotic therapy. The response tovarious combinations of treatment in this series was variable.

Oral prednisolone was given for 6 weeks at an initial dose of 60mg daily in divided doses, which were tapered slowly with clinicalimprovement. Of the 12 patients who received steroid therapy, 3patients had full-blown recurrent diseases and 3 others complainedof non-tender induration after their first course of steroid treat-ment (recurrence rate 6/12 = 50.0%).

Of the 2 patients with recurrent breast abscesses, one patientdeveloped recurrence 8 months after a short 4-week course of ste-roid therapy. Nonetheless the recurrent disease responded well to asecond course of steroid therapy. The other patient developed re-currence after concurrent treatment with antibiotics and steroids atthe time when the wound had not completely healed. Both had an-other drainage and a second course of steroid therapy with goodresponse. The three patients who complained of non-tender indu-ration after a 6-week course of steroid therapy also completed asecond course of steroid therapy, after which their induration sub-sided. In this series the complete response to the treatment ofCGM varied from 6 weeks to 11 months. The steroid dosage of

prednisolone 20 mg three time daily was prescribed for 2 weeks andin a tailing dosage over 6 weeks depending on the clinical response.Patients who had recurrences were continued on low-dose prednis-olone until complete clinical response were observed. In contrast,those who had secondary infection and abscess formation neededmore time to heal. Tables 1 and 2 illustrate the data on manage-ment and response of patients in this series with the clinical impres-sion of abscess and suspicious breast lesions. The response to ste-roid treatment was variable.

Discussion

Breast cancer continues to be the most common cancer among Ma-laysian women. The strong negative social-cultural perception ofthe disease together with remote location in rural areas and inac-cessibility to health care combine to account for the delayed pre-sentation and the advanced stage of disease at presentation. At thesame time, a misdiagnosis of breast carcinoma can easily be madein patients with chronic granulomatous mastitis.

Chronic granulomatous mastitis is a rare benign breast diseasecondition characterised by a chronic necrotizing granulomatouscondition. Although the origin remains unknown, it has been pos-tulated that CGM could result from an autoimmune localized re-sponse to the retained and extravasated fat- and protein-rich secre-tions in the duct [3]. It is said to occur predominantly in women ofchildbearing age. The mean age of patients with CGM in this studywas 36.5 years, which was comparable to that in previous studies.Most of these women were diagnosed within five years of their lastchildbirth. In addition, there appears to be no association withbreast-feeding, smoking, or any hormonal treatment.

Chronic granulomatous mastitis may manifest itself in two ways:first, as an abscess and second, mimicking a breast carcinoma. Inthis study, all patients presented with palpable breast lesions, whichoften involved a whole quadrant of the breast. The lesions are usu-ally unilateral although there have been reported cases of bilateralbreast involvement. There appears to be no tendency for the lesionto occur on particular side. The diagnosis of CGM remains a diag-nosis of exclusion. Although it can mimic breast cancer clinicallyand radiologically, neither mammography (Fig. 2) nor ultrasonog-raphy is said to play a significant role in the differential diagnosis of

Fig. 1. Chronic granulomatous mastitis.

Table 1. Management and response of patients with initial clinicalimpression of breast abscess.

TreatmentFrequency(n = 12) Response (follow-up 2 months)

Drainage plus antibiotics 8/12 NilDrainage plus antibiotics

with steroids4/12 Recurrent abscess (n = 2)

Induration (n = 1)

Table 2. Management and response of patients with initial clinicalimpression of suspicious breast lesion.

TreatmentFrequency(n = 13)

Response (follow-up6 months)

Steroid only 7/13 Recurrence (n = 1)Steroid + antibiotics 3/13 Induration (n = 1)Excision biopsy + antibiotics

+ steroids3/13 Induration (n = 1)

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granulomatous mastitis [4]. Gray-scale ultrasonography has beenreported to show findings specific for granulomatous mastitis insome cases, but all cases still require histopathologic confirmationfor diagnosis [5–7]. In this study, the definitive diagnosis was onlymade through histopathological findings either from an excisionbiopsy or core needle biopsy. Exclusion of other known causes ofgranulomatous inflammations should be performed such as fungal,foreign body, or acid-fast bacilli. Although other autoimmunegranulomatous breast conditions were also taken into consider-ation, we felt that further detailed investigations for this conditionwere not warranted as none of these patients complained of anysymptoms or had a family history that might suggest these causes.

Microscopically, CGM may appear with foamy macrophages andmultinucleated giant cells forming granulomas and microabscesses(Fig. 1). The ducts may appear normal and there is no evidence ofmalignancy. Stains for fungi and acid-fast bacilli are negative. Eachcourse of steroid therapy lasted for 6 weeks with an initial dose of 60mg daily. Patients were followed up closely and the doses were ta-pered once the condition showed clinical improvement. Of our 12patients who received steroids, 6 showed resolution after a singlecourse of steroid therapy and 6 required two courses of steroids.Three of the 12 patients developed full-blown recurrent diseaseprobably due to insufficient exposure to steroids. Nevertheless, be-cause the recurrence developed 8 months after completion of thefirst course of steroid therapy, it is possible that CGM may lie dor-mant and, for unknown reasons, may recur months later. Thus,long-term follow-up is recommended in patients with chronicgranulomatous mastitis. It remains uncertain whether long-termlow-dose therapy may be offered as an additional measure to pre-vent recurrence.

Observation in the present study recommends excision biopsywhenever possible coupled with steroid therapy. In conditions com-plicated by abscesses, surgical drainage and antibiotic treatmentmust be advocated prior to starting steroid therapy. In this series,the use of steroids showed variable results and raises the possibilitythat perhaps the steroid therapy should be advocated at smallerdoses but for a longer duration.

In conclusion, chronic granulomatous mastitis is a rare benignbreast condition that may be misdiagnosed as breast carcinoma.Because clinical or imaging diagnosis has often been difficult andinconclusive, histopathology remains the optimal diagnostic tool.In this series, we recommend complete resection whenever pos-sible and steroid therapy as an adjuvant for optimal treatment. Be-cause about 50% of patients experience recurrences, long-term fol-low-up is necessary. Awareness among surgeons and pathologistsshould also be emphasized to avoid unnecessary misdiagnosis.

Résumé. Cette étude rétrospective revoit la présentation, les donnéeshistopathologiques et le traitement optimal de la mastite granulomateusechronique. Site. Hôpital universitaire, Malaisie. 25 femmes qui avaient lescritères histologiques nécessaires, entre jan. 1998 et déc. 2000. Critèresprincipaux. Morbidité et récidive de la maladie. Treize femmes seprésentaient avec une masse au sein simulant cliniquement un cancer et 12patientes avaient une induration du sein avec abcès. Huit patientes seprésentaient avec un cancer du sein récidivé. Le diagnostic clinique etradiologique est souvent difficile et non concluant. Cependant, l’examenhistopathologique reste l’outil diagnostique optimal. Ce qu’il faut retenirest que 50% des patients ont une récidivé et, par conséquent, qu’unesurveillance á long terme est nécessaire. La mastite granulomateusechronique est une maladie du sein rare qui peut simuler le cancer du sein.Nous recommandons pour un traitement optimal la résection complètechaque fois que possible et une corticothérapie comme traitementadjuvant. Un indexe de suspicion élevé parmi les chirurgiens et lesanatomopathologistes est à souligner pour ne pas méconnaître lediagnostic et y porter un traitement adapté.

Resumen. Trabajo retrospectivo para revisar la clínica, hallazgoshistopatológicos y tratamiento óptimo de la mastitis granulomatosacrónica. Lugar. Hospital docente de Malasia. Casuística. 25 mujeres quereunían los criterios histológicos requeridos tratadas entre enero de 1998 ydiciembre del 2000. Principales resultados incluyendo la morbilidad yrecidiva de la enfermedad. 13 pacientes presentaron una tumoraciónmamaria que clínicamente se asemejaba a un cáncer; 12 enfermasmostraron induración mamaria y abscesos; 8 casos eran recidivas. Eldiagnóstico tanto clínico como radiológico fue con frecuencia difícil y noconcluyente. El estudio histopatológico constituye el método idóneo para eldiagnóstico. En el 50% de los casos se producen recidivas por lo que seprecisa un largo seguimiento de estas pacientes. La mastitis granulomatosacrónica es una rara enfermedad benigna de la mama. El diagnósticodiferencial ha de plantearse sobre todo con el cáncer de mama. Comotratamiento óptimo recomendamos, la extirpación a ser posible junto a untratamiento adyuvante con esteroides. Esta afección ha de ser tenida muyen cuenta por los patólogos y cirujanos, para evitar errores en eldiagnóstico y en el tratamiento.

Acknowledgment

The authors thank the Director-General of Health Services, Ma-laysia, for his kind permission to publish this article.

References

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Fig. 2. Mammogram showing extensive microcalcification in a patient di-agnosed with chronic granulomatous mastitis, which may mimic the appear-ance of breast cancer.

517Azlina et al.: Chronic Granulomatous Mastitis

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