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Oral carcinoma in pregnancy
Oral carcinoma S. A.
Layton.
LIepurtment
M.
in pregnancy
Rintoul,
of Oral
U. S. Avery
and Maxillofacial Surgery,
Middlesbroqh
Ckwral
Hospiral, Middleshrough
SUMMARY. Oral cancer presenting in pregnancy is a rare combination which, because of current epidemiological changes in the disease, may present more often in the future. Treatment of the condition can be very difficult. Decisions have to be made about terminating the pregnancy or finding a balance between the treatment of the mother and protL&ing the foetus from the effects of various treatment modalities. This may result in a compromise treatment of the disease. To illustrate the problems, we discuss a case of carcinoma of the tongue presenting in a 23-week pregnant Gravida 1 Yara 0 lady, and review the literature.
INTRODUCTION Oral cancer is an uncommon
malignancy and whilst figures for the year 1980 (Parkin et al., 19X8) showed it to be the fourth commonest cancer in men, it was only the eighth commonest form of cancer in women with approximately half the incidence of men. Until recently it was thought that oral cancer was dccreasing in Incidence in western populations (Svcjda & Kosut. 1971) although a study by Szpack et ml. (1977) suggested that the decrease in incidence was only in men, accounting for a fall in the malc:fcmalc ratio of the disease. More recent research has shown that the incidence of oral cancer is increasing in both sexes and the trend is being reversed (Hakulincn ef al., 1586). There is also evidcncc to suggest that there is an increasing proportion of female patients developing carcinoma of the tongue (Gallery it al., 1984). Traditionally oral cancer has been a disease of the older population with 98% of patients presenting over the age of 40 years. but there is now cvidcnce to suggest that there is an increase in the disease affecting younger patients (Boyle et al., IWO). Therefore, although a young woman of childbearing age is unlikely to be a victim of oral cancer. recent changes in the epidemiology of oral cancer would suggest that the risk is increasing slightly. although this is difficult to substantiate. It must also be remembered that women arc having children by natural and artificial methods increasingly later in life. It is not unusual for the professional woman to start her family in her late thirties or early forties. bringing her into the ‘at risk’ age group for oral cancer. The
patient, her partner and the clinician must address several difficult decisions when oral cancer presents in pregnancy. First to bc considered is the question of the possibility of immediate termination of pregnancy which of course would eliminate this complicating factor. If the pregnancy is to bc continued problem< arise because of the risk to the
foetus from various modalities of treatment. It may be that the ideal treatment for the type. site and stage of tumour may potentially offer an unacceptably high risk to the foetus or outcome of the pregnancy. In these circumstances a compromise treatment plan may have to bc implemented. WC present details of a patient recently under the care of one of the authors (BSA). WC do not suggest that any conclusions can be drawn from one case. Nevertheless the surgeon dealing with oral malignancy is unlikely to see more than one case in his or her career and the management presents a multitude of clinical, moral and emotional problems for all concerned. As such it may be valuable to document and discuss the presentation. management and outcome of one such patient and to review the available literature.
Case report A 27-year-old
female was referred to the Oral and Maxillobrcial Surgery Department compl;lining of a white patch on the left side of her tongue which was occasionally painFul. Her medical history was clear apart from the fact that she was 15 weeks pregnant (Gravida I Para 0). She had smoked approximately 10 cigarettes a dily from her early teenage years until a few weeks before prcscntation. Examination rcvealcd reticular pattern white patches on her right and left buccal mucosa and the Icft lateral border of tongue. There was no Iyn~padcnopath~ and no cutaneous Icsions were present. A provisional diagnosis of lichen planus was made and a follow-up appointment planned after the birth of her baby. The patient rcprescntcd 2 months later complaining ol recurrent discomfort from her tongue as well x pain in her left car and throat. Examination now showed a definite
swelling of approximately laterA border of tongue.
2.2 cm diamctcr
on the left
with surrounding kcratosis. Incisional biopsy. under local anaesthctic. was pcrformcd and histological examination showed poorly differentiated squanious cell carcinoma deeply invading muscle. An cnlqcd upper left decpccrvical lymph node was p:llp:rblc. Stqing of the tumour was difficult without knowing the
162
British Journal of Oral and Maxillofacial
Surgery
extent of the invasion of the tongue musculature and the nature of the enlarged lymph node in the neck. At this time it was Iabellcd a ‘I‘2 tumour with possible ipsilateral single node involvement. therefore cithcr Stage 2 or 3. After extensive discussion with the patient and her partner, termination of pregnancy was rejected by them as an option. Major surgery was excluded because of the risk to the foctus, as was interstitial radiotherapy. The appropriate trcatmcnt was therefore considered to bc a radical course of external beam radiotherapy to the patient’s tongue and left upper neck, with the appropriate protection of the abdomen and pelvis with lead shields. The maximum tumour dose of 60 Gray was given in 30 fractions over a 6week period. Considerable thought was given to the amount of radiation received by the unborn child. At the beginning of the radiotherapy treatment, measurements of the radiation dose to the abdomen were carried out. According to these, for the entire course of treatment. it was estimated the foetus would receive approximately 15.9 c Gy exposure. This would increase the risk of developing childhood cancer, but given in the second trimester of pregnancy should not be associated with an increased risk of foctal malformation. Radiotherapy was completed shortly before the delivery of the baby which was induced at 36 weeks. The vaginal delivery was uncomplicated and the child was healthy at birth. Five weeks. after the completion of the radical radiotherapy. the tongue tumour had regressed leaving an arca of scarring and induration. The cervical lymph node had reduced in size leaving a small residual mass which was to be closely monitored. Unfortunately 3 months later the patient developed a recurrence of the tongue tumour. Salvage surgery was performed consisting of tracheostomy. left radical neck dissection, lip and mandible split, left hcmiglossectomy and partial mandibulcctomy from premolar to angle region. Reconstruction was by means of a left pectoralis major myocutaneous flap. She made a good recovery and completed a course of hyperbaric oxygen therapy to decrease the risk of osteoradionecrosis. Approximately I year later reconstruction of the mandible was carried out with a Dacron tray and iliac crest bone graft which restored mandibular continuity, with subsequent good bone formation. Hyperbaric oxygen therapy was also used in association with this surgery. Twenty months after initial presentation the patient attended complaining of left side facial pain. A cystic lesion was present in the left fauccs region. This was biopsied and histological examination confirmed the presence of poorly differentiated squamous cell carcinoma. Further surgery or radiotherapy wcrc considered inappropriate at this stage and the patient refused chemotherapy. She died of local disease with no clinical evidence of mctastascs, 2 years after the initial presentation. No postmortem was performed at the request of her family. DISCUSSION
There is no evidence to suggest that pregnancy 1~” SE affects the course or outcome of oral malignancy and indeed the prognosis of many different types of malignancy has been shown to bc unrelated to pregnancy (Bechcr & Ilyman, 195.5; Crcasman er al.. 1970: Doncgan. 1977). However the coexistence of pregnancy may mean either compromising treatment to minimisc harm to the foetus, or altcrnativcly providing optimum treatment with consequent
unacceptable risk to the foetus. It may therefore be prudent to consider termination of pregnancy as the initial ‘treatment of oral cancer presenting in early pregnancy, thereby removing a complicating factor. Whether this is considered. let alone offered, is dependent on many factors and is such a compicx moral and ethical dilemma that it would be inappropriate to offer definitive views in this paper.
It has been suggested that in the case of pregnant patients with Hodgkins disease treatment should be postponed and the patient observed closely until after delivery of the baby (Jacob et al., 1981). The natural history of oral carcinoma precludes such a protocol except when it presents at a stage of pregnancy when it is considered obstetrically safe to induce delivery or where such a time is very close. How long one would be prepared to wait may depend on several factors including the histopathology, site and stage of disease at presentation. In the case described here, a carcinoma of the tongue presented in a young woman late in the second trimester of pregnancy. The possibility of termination of pregnancy was discussed and rejected by the parents. The lesion was T2NOMO-stage 2 disease-with the possibility of ipsilateral single node involvement which would make it stage 3 disease. The management of tongue carcinoma, especially where the staging of the disease is unclear, is controversial. Single modality treatment, either surgery or radiotherapy, is generally considered adequate for early stage 1 and 2 oral squamous carcinomas. However, it has been suggested that single modality treatment in the form of radiotherapy is probably inappropriate and inadequate in treating most carcinomas of the tongue and surgery with postoperative radiotherapy is the optimum treatment. Additionally because the likelihood of regional lymph node involvement is so high in tongue tumours, surgical treatment of the neck should form part of the initial management of most 12 lesions and all more advanced lesions of the tongue (Strong, 1990). In keeping with this current thinking about the management of tongue carcinoma, the treatment of choice of the author (BSA), who was the surgeon treating this case, would have been surgical treatment of the primary tumour including radical neck dissection. Postoperative radiotherapy, however, would have been used only if multiple nodal metastasis or extra-capsular spread were demonstrated. Radiotherapy alone would be a poor second choice in a case such as this one and would only be considered acceptable as a single modality treatment if surgery was contraindicated as in this case. Interstitial radiotherapy was not considered appropriate for several reasons. Firstly, in the opinion of the radiotherapist. the size of the lesion was at the upper limit of suitability for interstitial therapy. Interstitial implants would require an operative procedure, ideally under general anacsthesia. and it was considered this would bc an unacceptable risk to the foctus. Additionally, the presence of an enlarged cervical lymph node and the likelihood of regional lymph node involvement in carcinoma of the tongue
suggested that external beam radiation to the primary tumour and neck was the best compromise treatment for this patient. Estimates were made however of foctal exposure to radiation with both interstitial and external beam radiotherapy and were calculated at around 7 cG for both, although calculations based on measurements made during treatment showed this was probably an undercstimatc for external beam radiotherapy. Not only is external beam radiotherapy not the ideal primary treatment in this case, it is associated vvith two significant risks which warrant mention. Firstly there is the possibility of radionccrosis, particularly ostcoradionccrosis of the mandible. hence the use of hyperbaric oxygen therapy with surgery in this patient. Secondly there is the long term risk of radiation induced neoplasia and the development of a second primary tumour in the area of irradiation (Lawson & Som, lY75). a point that is particularly relevant in a younger patient. Whether the tumour which dcvelopcd after surgical treatment and led to the demise of our patient was a recurrence or second primary is not possible to say. Although the initial resection was reported histologically as having clear margins, the histological similarity suggests that the patient suffered a recurrence rather than a second tumour. The main risk to the foctus of exposure to radiation during the first 17 weeks of pregnancy is malformation, which in our case is not relevant. Exposure to radiation during the last 7 months of pregnancy increases the risk of childhood cancer, which was of relevance to our patient and her child. The natural risk of childhood cancer is 100~ 10-s (1 in a thousand) and the risk due to radiation exposure during the last 7 months of pregnancy is calculated to be 2.3x10-s per 0.1&y (Wong et al., 1986). The risk in our case of radiation-induced childhood cancer due to foetal exposure is therefore 159~2.3~ IO-” which yields a total risk of I(lOXlt)-’ (natural risk) plus 366~ 10m ’ (radiation induced risk)=466X IO- (4.66 in a thousand). Thcreforc, because of the exposure to radiation during pregnancy, it is likely in this case that the risk of childhood cancer is approximately 4-5 times normal. Reports of oral carcinoma in pregnancy arc very rare and this is true for all tumours of the head and neck. Shibuva cf uf. (lY87) described two cases of carcinoma of the tongue (stage 2) presenting during pregnancy in the 26th and 28th week, treated with external beam radiotherapy immediately and interstitial radiotherapy after the successful end of the pregnancy. One patient required subsequent neck dissection, but both were free of disease at 4 years at time of publication. In our patient, external beam radiotherapy did not control the tumour which recurred and required salvage surgery after the successful birth of her, to date, healthy baby. Unfortunately this surgery was also unsuccessful in the medium term due to the recurrence of tumour. Ferlito and Nicolai (1980) in discussing treatment of laryngeal carcinoma in pregnancy strongly criticised the USC of radiotherapy as a compromise because of
pregnancy and said that the treatment of choice for such a condition in a young person was surgery which should be pursued regardless of the state of pregnancy. They did not say whether they advocated preliminary termination of pregnancy or whether they advocated surgery on the pregnant woman. as does Brophy (1973). Brophy (1973) described a case of laryngeal carcinoma operated on by hemilaryngcctomy during the midtrimcstcr of pregnancy. At all stages of pregnancy. the foctus is at risk from one or more aspect of surgery and anaesthesia. In the first trimester of pregnancy the teratogenic effects of drugs and spontaneous abortion arc the most severe problems. Throughout the whole pregnancy the foctus is at risk from hypoxaemia and in the third trimester prcmaturc labour may result from anaesthesia and surgery. The middle trimester is relatively the safest period in which to carry out surgery m pregnancy, but still involves considerable risk to the foetus. Although not relevant in our case because of the riced for treatment of the neck as discussed earlier, moderate surgery such as local resection of stage I and small stage 2 oral tumours in the second trimester of pregnancy may offer both optimum trcatmcnt combined with minimal obstetric risk. Fortunately the chances of a surgeon seeing a case of oral carcinoma in pregnancy is extremely small. although there is a possibility it is becoming more likely. However the rarity of the condition means it is impossible to acquire any experience and equally difficult to accumulate any collective data from different ccntrcs. There exists no treatment protocol and it is not the intention of this paper to suggest enc. Essentially two alternative treatment outlines exist. Firstly the child can be deliberately aborted or placed at risk by major surgery during pregnancy. This has to be considered in the face of very poor survival figures for this disease even with optimum treatment. Secondly the patient can bc compromised by minimising the risk to the foetus by using a less than ideal treatment. No treatment modality is entirely free of immediate or delayed risk to the foetus and only the magnitude of risk is rcduccd. Just which road a clinician takes with the patient is a collective decision based on many complex medical. social, moral, ethical and emotional factors.
Acknowledgements The authors thank Dr J. I‘. Roberts. Consultanr Kadiothrrapisl and Oncologist. for hiz advice during the preparation of this paplX.
References l3cchur.M. D.&Hgrn;~n.<.~.A.( lYh~)Mnnagcmcnto~~lodgkins diseltsccocxistcntwithprcpnancy. Rudiolo~y. 85.725. Roylc. P.. Macfarl;lne.<;. J . Maisonncwc. P.. Zhcr1g.T.. Scull_v. C. &l‘cdesco. H. ( 1990). Epidcmiolo~yofmouth cancer in 1980: arevciw. Joundo/‘~hc RuwISorir~.vo/ Mediciuc. 83.724. l3rophy.J. W.( 1973). Squamous~c;lrcinomaofthclar).nxirl pregnancy. ,1rc./rive~c!l’O/o/ary,rRoloR)‘. 97,3X0.
I64
British
Journal
of Oral
and MaxilloPacial
Surgery
Callery,C~.I~..Spiro.R.II.&Strong.E.WW.(I984).Changing trcndsinthetnanagcmcntofcarcinomaofthctonguc. American Journal[lffSLtr~sc,rr, 148,449 Crcasman. W.‘I‘.. Rutledge. F. N.&Fletcher. G. H. (1070). Carcinomaofthccervixassociatcdwithprcgnancy. OhslerrirsaflrlG~r~ecolo~~. 36,4YS. D0ncgan.W. I.. (lY77). Hrcastcancerandpregnancy. Ohs/~trics trnrl Gjwwlog~. 50,244. Ferlito.A.&Nicolal.P.(1980). Laryngealcancerinpregnancy Acrcr Olorlrinolar~~olica Belgica, 34,706. I-lakulincn.T..Andcrsen.A. A.. Ma1ker.B.. Pukkala. E.. Schou.Ci.&l‘ulinius.ll. (IYXh).l‘rcndsincancerincidcncc in the NordicCountrics. Acra fulho/o,qicu, Microhio/o,qica. Scundanctvicn. Supplement. 28X. I. J;lcob.C‘..Donaldson.S.S..Roscnbere.S.A.&Kaolan.ll.S (IYXI). Managcmentofprc~n;lntI;atientwithIiodgkins disease. A~~n~/.so.//n/~~r~~cr/Madicine. 95,660. Lawson. W.BSom.M.( 1975).Sccondprimarycanccraftcr Rhinolog): irradiationo~larynge;~lcanccr.Annu/.sqfO~olo~~~. and I.arygology. X4.77 I. Parkin,D.~l..L;~ara.E.&Muir.C.S.(IYX8).Estimatcsolthe worldwide Itequencvofsixtccnmaiorcanccrsin 1980. I,rrcrnatio,~alJo~cr~~~~~~~f C‘ancer. 41, I X4. Shibuva. H..Saio\. M.. Horiuchi. J. I. & Suzuki.S. C19x7). l’rcatmcnt ofmalignant hcadand neck tumouriduring prqnancy-a rcportof3cascs. AcraOnrolo~icu. 26,237. Strong. E. ( 1900). Surgical management oforal cancer. Derfrcrl C%',licalojMorrh Americu. 34. 185. Svejda.J.XcKosut.V.( 1971). EpidcmioloRyolm;llignant
tumours\r,lthspccial regardtotheorofacialregion. Ncoplasmu. IX, 193. Szpack.C. A..Stonc.J.J.&Frenkcl.E.P.(1977).Some ohservationsconcerningthcdcmogr~lphicandgeoeraphic incidcnceofc;lrcinomaofthc lipand huccalcavity. Cancer. 40,343. Wong.F..Sai-Ki.O..Chcung.F.&Schiu,W. (1986). Pregnancy outcome followingradiothcrap~fornaso-pharyngcal carcinoma. Europran Journalof Ohrfe,ric.s. Gpnaecologyand Kcproduclion Biology. 22,lS7.
The Authors S. A.
I.agton FDSRCS. FRCS Rcgislrar M. Rintoul RI.8 Senior I louse Officer B.S. Avery FDSRCS, FRCS Consultant DcpartmcntofOralandMaxillofacialSurgcry MiddlcsbroughGcneralHospital AyresomcGrccn Lane Middleshroughl‘S5SAZ CorrcspondcnceandrequcstsforolfprintstoMrS. Paperrcccivcd22 August 1991 Accepted9 Dccembcr 1991
A.Layton
Layton.
LIepurtment
M.
in pregnancy
Rintoul,
of Oral
U. S. Avery
and Maxillofacial Surgery,
Middlesbroqh
Ckwral
Hospiral, Middleshrough
SUMMARY. Oral cancer presenting in pregnancy is a rare combination which, because of current epidemiological changes in the disease, may present more often in the future. Treatment of the condition can be very difficult. Decisions have to be made about terminating the pregnancy or finding a balance between the treatment of the mother and protL&ing the foetus from the effects of various treatment modalities. This may result in a compromise treatment of the disease. To illustrate the problems, we discuss a case of carcinoma of the tongue presenting in a 23-week pregnant Gravida 1 Yara 0 lady, and review the literature.
INTRODUCTION Oral cancer is an uncommon
malignancy and whilst figures for the year 1980 (Parkin et al., 19X8) showed it to be the fourth commonest cancer in men, it was only the eighth commonest form of cancer in women with approximately half the incidence of men. Until recently it was thought that oral cancer was dccreasing in Incidence in western populations (Svcjda & Kosut. 1971) although a study by Szpack et ml. (1977) suggested that the decrease in incidence was only in men, accounting for a fall in the malc:fcmalc ratio of the disease. More recent research has shown that the incidence of oral cancer is increasing in both sexes and the trend is being reversed (Hakulincn ef al., 1586). There is also evidcncc to suggest that there is an increasing proportion of female patients developing carcinoma of the tongue (Gallery it al., 1984). Traditionally oral cancer has been a disease of the older population with 98% of patients presenting over the age of 40 years. but there is now cvidcnce to suggest that there is an increase in the disease affecting younger patients (Boyle et al., IWO). Therefore, although a young woman of childbearing age is unlikely to be a victim of oral cancer. recent changes in the epidemiology of oral cancer would suggest that the risk is increasing slightly. although this is difficult to substantiate. It must also be remembered that women arc having children by natural and artificial methods increasingly later in life. It is not unusual for the professional woman to start her family in her late thirties or early forties. bringing her into the ‘at risk’ age group for oral cancer. The
patient, her partner and the clinician must address several difficult decisions when oral cancer presents in pregnancy. First to bc considered is the question of the possibility of immediate termination of pregnancy which of course would eliminate this complicating factor. If the pregnancy is to bc continued problem< arise because of the risk to the
foetus from various modalities of treatment. It may be that the ideal treatment for the type. site and stage of tumour may potentially offer an unacceptably high risk to the foetus or outcome of the pregnancy. In these circumstances a compromise treatment plan may have to bc implemented. WC present details of a patient recently under the care of one of the authors (BSA). WC do not suggest that any conclusions can be drawn from one case. Nevertheless the surgeon dealing with oral malignancy is unlikely to see more than one case in his or her career and the management presents a multitude of clinical, moral and emotional problems for all concerned. As such it may be valuable to document and discuss the presentation. management and outcome of one such patient and to review the available literature.
Case report A 27-year-old
female was referred to the Oral and Maxillobrcial Surgery Department compl;lining of a white patch on the left side of her tongue which was occasionally painFul. Her medical history was clear apart from the fact that she was 15 weeks pregnant (Gravida I Para 0). She had smoked approximately 10 cigarettes a dily from her early teenage years until a few weeks before prcscntation. Examination rcvealcd reticular pattern white patches on her right and left buccal mucosa and the Icft lateral border of tongue. There was no Iyn~padcnopath~ and no cutaneous Icsions were present. A provisional diagnosis of lichen planus was made and a follow-up appointment planned after the birth of her baby. The patient rcprescntcd 2 months later complaining ol recurrent discomfort from her tongue as well x pain in her left car and throat. Examination now showed a definite
swelling of approximately laterA border of tongue.
2.2 cm diamctcr
on the left
with surrounding kcratosis. Incisional biopsy. under local anaesthctic. was pcrformcd and histological examination showed poorly differentiated squanious cell carcinoma deeply invading muscle. An cnlqcd upper left decpccrvical lymph node was p:llp:rblc. Stqing of the tumour was difficult without knowing the
162
British Journal of Oral and Maxillofacial
Surgery
extent of the invasion of the tongue musculature and the nature of the enlarged lymph node in the neck. At this time it was Iabellcd a ‘I‘2 tumour with possible ipsilateral single node involvement. therefore cithcr Stage 2 or 3. After extensive discussion with the patient and her partner, termination of pregnancy was rejected by them as an option. Major surgery was excluded because of the risk to the foctus, as was interstitial radiotherapy. The appropriate trcatmcnt was therefore considered to bc a radical course of external beam radiotherapy to the patient’s tongue and left upper neck, with the appropriate protection of the abdomen and pelvis with lead shields. The maximum tumour dose of 60 Gray was given in 30 fractions over a 6week period. Considerable thought was given to the amount of radiation received by the unborn child. At the beginning of the radiotherapy treatment, measurements of the radiation dose to the abdomen were carried out. According to these, for the entire course of treatment. it was estimated the foetus would receive approximately 15.9 c Gy exposure. This would increase the risk of developing childhood cancer, but given in the second trimester of pregnancy should not be associated with an increased risk of foctal malformation. Radiotherapy was completed shortly before the delivery of the baby which was induced at 36 weeks. The vaginal delivery was uncomplicated and the child was healthy at birth. Five weeks. after the completion of the radical radiotherapy. the tongue tumour had regressed leaving an arca of scarring and induration. The cervical lymph node had reduced in size leaving a small residual mass which was to be closely monitored. Unfortunately 3 months later the patient developed a recurrence of the tongue tumour. Salvage surgery was performed consisting of tracheostomy. left radical neck dissection, lip and mandible split, left hcmiglossectomy and partial mandibulcctomy from premolar to angle region. Reconstruction was by means of a left pectoralis major myocutaneous flap. She made a good recovery and completed a course of hyperbaric oxygen therapy to decrease the risk of osteoradionecrosis. Approximately I year later reconstruction of the mandible was carried out with a Dacron tray and iliac crest bone graft which restored mandibular continuity, with subsequent good bone formation. Hyperbaric oxygen therapy was also used in association with this surgery. Twenty months after initial presentation the patient attended complaining of left side facial pain. A cystic lesion was present in the left fauccs region. This was biopsied and histological examination confirmed the presence of poorly differentiated squamous cell carcinoma. Further surgery or radiotherapy wcrc considered inappropriate at this stage and the patient refused chemotherapy. She died of local disease with no clinical evidence of mctastascs, 2 years after the initial presentation. No postmortem was performed at the request of her family. DISCUSSION
There is no evidence to suggest that pregnancy 1~” SE affects the course or outcome of oral malignancy and indeed the prognosis of many different types of malignancy has been shown to bc unrelated to pregnancy (Bechcr & Ilyman, 195.5; Crcasman er al.. 1970: Doncgan. 1977). However the coexistence of pregnancy may mean either compromising treatment to minimisc harm to the foetus, or altcrnativcly providing optimum treatment with consequent
unacceptable risk to the foetus. It may therefore be prudent to consider termination of pregnancy as the initial ‘treatment of oral cancer presenting in early pregnancy, thereby removing a complicating factor. Whether this is considered. let alone offered, is dependent on many factors and is such a compicx moral and ethical dilemma that it would be inappropriate to offer definitive views in this paper.
It has been suggested that in the case of pregnant patients with Hodgkins disease treatment should be postponed and the patient observed closely until after delivery of the baby (Jacob et al., 1981). The natural history of oral carcinoma precludes such a protocol except when it presents at a stage of pregnancy when it is considered obstetrically safe to induce delivery or where such a time is very close. How long one would be prepared to wait may depend on several factors including the histopathology, site and stage of disease at presentation. In the case described here, a carcinoma of the tongue presented in a young woman late in the second trimester of pregnancy. The possibility of termination of pregnancy was discussed and rejected by the parents. The lesion was T2NOMO-stage 2 disease-with the possibility of ipsilateral single node involvement which would make it stage 3 disease. The management of tongue carcinoma, especially where the staging of the disease is unclear, is controversial. Single modality treatment, either surgery or radiotherapy, is generally considered adequate for early stage 1 and 2 oral squamous carcinomas. However, it has been suggested that single modality treatment in the form of radiotherapy is probably inappropriate and inadequate in treating most carcinomas of the tongue and surgery with postoperative radiotherapy is the optimum treatment. Additionally because the likelihood of regional lymph node involvement is so high in tongue tumours, surgical treatment of the neck should form part of the initial management of most 12 lesions and all more advanced lesions of the tongue (Strong, 1990). In keeping with this current thinking about the management of tongue carcinoma, the treatment of choice of the author (BSA), who was the surgeon treating this case, would have been surgical treatment of the primary tumour including radical neck dissection. Postoperative radiotherapy, however, would have been used only if multiple nodal metastasis or extra-capsular spread were demonstrated. Radiotherapy alone would be a poor second choice in a case such as this one and would only be considered acceptable as a single modality treatment if surgery was contraindicated as in this case. Interstitial radiotherapy was not considered appropriate for several reasons. Firstly, in the opinion of the radiotherapist. the size of the lesion was at the upper limit of suitability for interstitial therapy. Interstitial implants would require an operative procedure, ideally under general anacsthesia. and it was considered this would bc an unacceptable risk to the foctus. Additionally, the presence of an enlarged cervical lymph node and the likelihood of regional lymph node involvement in carcinoma of the tongue
suggested that external beam radiation to the primary tumour and neck was the best compromise treatment for this patient. Estimates were made however of foctal exposure to radiation with both interstitial and external beam radiotherapy and were calculated at around 7 cG for both, although calculations based on measurements made during treatment showed this was probably an undercstimatc for external beam radiotherapy. Not only is external beam radiotherapy not the ideal primary treatment in this case, it is associated vvith two significant risks which warrant mention. Firstly there is the possibility of radionccrosis, particularly ostcoradionccrosis of the mandible. hence the use of hyperbaric oxygen therapy with surgery in this patient. Secondly there is the long term risk of radiation induced neoplasia and the development of a second primary tumour in the area of irradiation (Lawson & Som, lY75). a point that is particularly relevant in a younger patient. Whether the tumour which dcvelopcd after surgical treatment and led to the demise of our patient was a recurrence or second primary is not possible to say. Although the initial resection was reported histologically as having clear margins, the histological similarity suggests that the patient suffered a recurrence rather than a second tumour. The main risk to the foctus of exposure to radiation during the first 17 weeks of pregnancy is malformation, which in our case is not relevant. Exposure to radiation during the last 7 months of pregnancy increases the risk of childhood cancer, which was of relevance to our patient and her child. The natural risk of childhood cancer is 100~ 10-s (1 in a thousand) and the risk due to radiation exposure during the last 7 months of pregnancy is calculated to be 2.3x10-s per 0.1&y (Wong et al., 1986). The risk in our case of radiation-induced childhood cancer due to foetal exposure is therefore 159~2.3~ IO-” which yields a total risk of I(lOXlt)-’ (natural risk) plus 366~ 10m ’ (radiation induced risk)=466X IO- (4.66 in a thousand). Thcreforc, because of the exposure to radiation during pregnancy, it is likely in this case that the risk of childhood cancer is approximately 4-5 times normal. Reports of oral carcinoma in pregnancy arc very rare and this is true for all tumours of the head and neck. Shibuva cf uf. (lY87) described two cases of carcinoma of the tongue (stage 2) presenting during pregnancy in the 26th and 28th week, treated with external beam radiotherapy immediately and interstitial radiotherapy after the successful end of the pregnancy. One patient required subsequent neck dissection, but both were free of disease at 4 years at time of publication. In our patient, external beam radiotherapy did not control the tumour which recurred and required salvage surgery after the successful birth of her, to date, healthy baby. Unfortunately this surgery was also unsuccessful in the medium term due to the recurrence of tumour. Ferlito and Nicolai (1980) in discussing treatment of laryngeal carcinoma in pregnancy strongly criticised the USC of radiotherapy as a compromise because of
pregnancy and said that the treatment of choice for such a condition in a young person was surgery which should be pursued regardless of the state of pregnancy. They did not say whether they advocated preliminary termination of pregnancy or whether they advocated surgery on the pregnant woman. as does Brophy (1973). Brophy (1973) described a case of laryngeal carcinoma operated on by hemilaryngcctomy during the midtrimcstcr of pregnancy. At all stages of pregnancy. the foctus is at risk from one or more aspect of surgery and anaesthesia. In the first trimester of pregnancy the teratogenic effects of drugs and spontaneous abortion arc the most severe problems. Throughout the whole pregnancy the foctus is at risk from hypoxaemia and in the third trimester prcmaturc labour may result from anaesthesia and surgery. The middle trimester is relatively the safest period in which to carry out surgery m pregnancy, but still involves considerable risk to the foetus. Although not relevant in our case because of the riced for treatment of the neck as discussed earlier, moderate surgery such as local resection of stage I and small stage 2 oral tumours in the second trimester of pregnancy may offer both optimum trcatmcnt combined with minimal obstetric risk. Fortunately the chances of a surgeon seeing a case of oral carcinoma in pregnancy is extremely small. although there is a possibility it is becoming more likely. However the rarity of the condition means it is impossible to acquire any experience and equally difficult to accumulate any collective data from different ccntrcs. There exists no treatment protocol and it is not the intention of this paper to suggest enc. Essentially two alternative treatment outlines exist. Firstly the child can be deliberately aborted or placed at risk by major surgery during pregnancy. This has to be considered in the face of very poor survival figures for this disease even with optimum treatment. Secondly the patient can bc compromised by minimising the risk to the foetus by using a less than ideal treatment. No treatment modality is entirely free of immediate or delayed risk to the foetus and only the magnitude of risk is rcduccd. Just which road a clinician takes with the patient is a collective decision based on many complex medical. social, moral, ethical and emotional factors.
Acknowledgements The authors thank Dr J. I‘. Roberts. Consultanr Kadiothrrapisl and Oncologist. for hiz advice during the preparation of this paplX.
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The Authors S. A.
I.agton FDSRCS. FRCS Rcgislrar M. Rintoul RI.8 Senior I louse Officer B.S. Avery FDSRCS, FRCS Consultant DcpartmcntofOralandMaxillofacialSurgcry MiddlcsbroughGcneralHospital AyresomcGrccn Lane Middleshroughl‘S5SAZ CorrcspondcnceandrequcstsforolfprintstoMrS. Paperrcccivcd22 August 1991 Accepted9 Dccembcr 1991
A.Layton